Social, Cultural Environmental Contexts

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Social, Cultural Environmental Contexts and the Measurement of the Burden of Disease

AN EXPLORATORY COMPARISON IN THE DEVELOPED AND DEVELOPING WORLD

Daniel D. Reidpath Pascale Allotey Aka Kouamé Robert Cummins

Social, Cultural and Environmental Contexts and the Measurement of the Burden of Disease. AN EXPLORATORY COMPARISON IN THE DEVELOPED AND DEVELOPING WORLD

Daniel D. Reidpath Pascale Allotey Aka Kouame Robert A. Cummins

March 2001

Funding Agencies: Global Forum for Health Research The University of Melbourne (MRCEG Scheme)

COPYRIGHT © 2001 BY DANIEL REIDPATH1, PASCALE ALLOTEY2, AKA KOUAMÉ3 & ROBERT CUMMINS4

1.

School of Health Sciences Deakin University Australia

2.

Key Centre for Women's Health in Society School of Population Health University of Melbourne Australia

3.

Institut de Formation et de Recherche Demographiques Cameroun

4.

School of Psychology Deakin University Australia

It is expected that the data on which this report is based will be available on the web site http://www.BurdenOfDisease.org from January 2002. Electronic copies of this report and the research protocols are available from the authors.

Suggested citation: Reidpath DD, Allotey P, Kouamé A & Cummins RA. 2001. Social cultural and environmental contexts and the measurement of burden of disease: An exploratory study in the developed and developing world. Melbourne: Key Centre for Women’s Health in Society, The University of Melbourne.

Any enquiries about or comments on this report should be directed to: Dr Daniel D Reidpath School of Health Sciences Deakin University 221 Burwood Highway Burwood Vic 3125 Australia Email: [email protected]

Any work of this nature will contain errors. We anticipate that none of them are serious. However, what errors there are, are ours alone. The views expressed are those of the authors and do not necessarily reflect the views of the affiliated institutions or the funding agencies.

Published by The Key Centre for Women’s Health in Society, Department of Public Health, School of Population Health, Faculty of Medicine, Dentistry and Health Sciences, The University of Melbourne.

Printed by Design and Print, The University of Melbourne.

FOREWORD (AUSTRALIA) Internationally, there is growing commitment to health policies and programs that are “evidence-based”: that is, that they derive from a body of research that has been proven true, effective or successful. In establishing an evidence-base, there is a continued concern with replicability of research, with the robustness of findings across time and place, and in the absence of replication, with an explanation for the lack of fit. In this endeavour, considerable attention has been paid to common tools, common protocols and consistent, shared measures – validated questionnaires, common tools to assess physical and mental health and summary indices for quality of life, health inputs and health outcomes. This concern for comparability in public health matches a concern by economists and health planners, locally, nationally and internationally, to rationalise, to set priorities and goals, to allocate funds on the objective basis of need and impact, and to direct resources where the outcome will be most effective. In this science and policy, the Disability Adjusted Life Year (DALY) offered a promising means to compute the cost of morbidity, to quantify pain, loss of function, impairment or handicap in order to summarise the impact on individuals of disease, accident, birth impediment or other change in function. The computation promised a means to compare internationally, as a result of which agencies and institutions – including government and Bretton Woods financing institutions such as the World Bank - might then adjust policies, flows of funds and the direction of other resources. The authors of this study (Reidpath, Allotey, Kouamé and Cummins) illustrate, however, that all is not equal, neither at the level of individual, community nor country. Social, economic and political circumstances, class, age and gender, family structure, support networks and community capacity – including what we refer to now as social capital – all influence personal response to illness, physical management and care, and psychological adjustment. They influence the ways in which family members too are able to accommodate disability, to share the costs of care, contribute to labour, meet the needs for medication and/or aids, and make up for loss of productivity when one householder’s contributions are reduced. While the DALY purportedly took on board such differences, and allowed for cross-country and cross-cultural comparison, this monograph – a study of paraplegia and epilepsy in Cameroon and Australia – makes clear how difficult it is to generalise, and how heavily context bears upon experience. The study is a provocative one. It highlights the subjective nature of disability, and the real differences that place, class and gender make. A young poor woman in rural Cameroon, crawling through mud to reach a latrine, has a very different experience of disability and its embodiment than does a woman of the same age in urban Australia, with a contemporary lightweight wheelchair and adjusted toilet facilities. The former relies on her family to feed her; in the absence of family, or her ability to beg. The latter lives in a society with a safety net for her security – the right to community care and case workers, assistance with aids, and minimal financial support to ensure she is not left to die. This monograph highlights the importance of studying different contexts and meanings of wellbeing, illness and disablement. There is a clear need for further thought to be given methodologically to and in terms of gathering the empirical evidence of disability. There is a wide variety of study techniques and approaches available to do this. Reidpath, Allotey, Kouamé and Cummins’ study highlights the value of combining qualitative and quantitative methods with the perspectives and insights of a variety of disciplines (in this case, anthropology, epidemiology and psychology) to document the variety of experiences of disability and illness. As health researchers, policy makers and the architects of programs, our ability to respond to illness, disease and dysfunction depends on the quality of the evidence-base. As this monograph illustrates, that base needs to be sensitive to context and to the complications that follow from life circumstances, opportunity and chance. Professor Lenore Manderson (Director) Key Centre for Women's Health in Society

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FOREWORD (CAMEROON) Health planners, public health specialists and demographers are trying to measure the health status of the population. In order to achieve this, they employ several indicators, particularly the mortality (infant and child), prevalence and incidence rates. However, these indicators have their own disadvantages. Concerning infant and child mortality, these measures became in mid 1980s a goal and an end. Consequently, it is common to fix policy/programme objectives in terms of a reduction in the rate of these indicators. Infant and child mortality rates have gradually become disassociated from the measurement of the health status of the population; and the emphasis has moved to children below 5 years. The launching of specific health programmes for children and the improvement of their health did not necessarily lead to improvement in the health of the whole population. It became necessary to develop indicators that could address the problem. The DALY (Disability Adjusted Life Year) is one of these instruments. It measures the combined impact of premature mortality and morbidity within a given population in terms of the number of years of life lost. Specifically, it measures the burden of disease in a society and is considered the basis for decision making in the health domain. However, this method has some shortcomings, in relation to the definition of disease severity. Despite its strong theoretical background and recognition, the DALY has never been confronted with empirical investigation. This study tries to verify one of the basic assumptions of the DALY according to which, the severity associated with a health condition does not vary according to location and socioeconomic conditions. In other words, the burden of the same disease does not vary according to country, place of residence and socioeconomic status of individuals. The authors therefore, decided to carry out a comparative study between Cameroon (in Central Africa) and Australia. These two countries present marked differences at the economic, cultural and political levels. As expected and contrary to the hypothesis underlying the DALY, the study shows that cultural and societal specificities should be taken into account in the determination of the severity weight that serve in the calculation of the DALY. Therefore the use of DALY as the basis for comparison should be used with caution. Apart from those methodological considerations, we are of the opinion that the DALY should be diffused to the scientific world, and particularly to the disciplines of the biomedicine, health and demography. The present work will pave the way for a wider diffusion of the method in Africa South of the Sahara. However, taking into consideration the masses and the quality of information required in order to apply appropriately the method, it may be very difficult to use the DALY on a large scale in Sub-Saharan African countries. Seemingly, in the short and medium terms, the use of this health indicator in Africa, will not be possible without foreign financial assistance. The partnership of Deakin University, the University of Melbourne and IFORD for taking the initiative to carry out this common project is highly appreciated in this regard. This first collaborative experience between these three institutions should be renewed and extended to other infections; as the social response of other diseases might be very different to that of epilepsy and paraplegia, which were the health conditions studied by this research. Professor AKOTO Eliwo (Director) Institut de Formation et de Recherche Demographiques

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TABLE OF CONTENTS AN EXPLORATORY COMPARISON IN THE DEVELOPED AND DEVELOPING WORLD.......................................................................................................................................... I FOREWORD (AUSTRALIA) ................................................................................................... III FOREWORD (CAMEROON) .................................................................................................. IV TABLE OF CONTENTS .............................................................................................................V ACKNOWLEDGEMENTS ......................................................................................................VII EXECUTIVE SUMMARY ..................................................................................................... VIII RÉSUMÉ ....................................................................................................................................XII CHAPTER 1 INTRODUCTION..................................................................................................1 MEASURES OF POPULATION HEALTH ..........................................................................................1 DEVELOPING THE DALY.............................................................................................................2 CRITICISMS OF THE DALY ..........................................................................................................3 THE GLOBAL SEVERITY WEIGHTS (GSW)...................................................................................3 DECONSTRUCTING THE GSW ......................................................................................................4 BACKGROUND TO THE GSW........................................................................................................4 THE “AVERAGE SOCIAL MILIEU”..................................................................................................5 MAKING GLOBAL COMPARISONS USING A GSW ..........................................................................6 QUESTIONS TO BE ANSWERED ....................................................................................................7 STRUCTURE .................................................................................................................................8 CHAPTER 2 METHODS ...........................................................................................................10 AIM ...........................................................................................................................................10 DESIGN ......................................................................................................................................10 COUNTRY ..................................................................................................................................10 GENDER .....................................................................................................................................12 ENVIRONMENT ..........................................................................................................................12 SOCIOECONOMIC STATUS ..........................................................................................................13 HEALTH CONDITIONS ................................................................................................................13 Epilepsy................................................................................................................................14 Paraplegia ...........................................................................................................................16 DATA COLLECTION ....................................................................................................................17 PHASE 1: INTERVIEW .................................................................................................................18 DATA ANALYSIS ........................................................................................................................19 ETHICS.......................................................................................................................................20 CHAPTER 3 RESULTS .............................................................................................................22 PARAPLEGIA ..............................................................................................................................22 PARAPLEGIA IN CAMEROON ......................................................................................................22 Gender factors in paraplegia in Cameroon .........................................................................23 Economic factors in paraplegia in Cameroon .....................................................................26 Environmental factors in paraplegia in Cameroon .............................................................27 Other social sequelae of paraplegia in Cameroon ..............................................................31 PARAPLEGIA IN AUSTRALIA.......................................................................................................32 Gender factors in paraplegia in Australia ...........................................................................33 Economic factors in paraplegia in Australia .......................................................................35 Environmental factors in paraplegia in Australia ...............................................................35 Other social sequelae of paraplegia in Australia ................................................................36 EPILEPSY ...................................................................................................................................37 EPILEPSY IN CAMEROON............................................................................................................37 Gender factors in epilepsy in Cameroon..............................................................................39

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Economic factors in epilepsy in Cameroon ......................................................................... 40 Environmental factors in epilepsy in Cameroon ................................................................. 41 Other social sequelae of epilepsy in Cameroon .................................................................. 41 EPILEPSY IN AUSTRALIA (VICTORIA) ........................................................................................ 41 Gender factors in epilepsy in Australia............................................................................... 43 Economic factors in epilepsy in Australia ........................................................................... 44 Environmental factors in epilepsy in Australia ................................................................... 45 Other social sequelae of epilepsy in Australia .................................................................... 46 QUANTITATIVE ANALYSIS .................................................................................................... 46 PARAPLEGIA ............................................................................................................................. 47 CAMEROON – QUANTITATIVE PARAPLEGIA DATA ..................................................................... 47 AUSTRALIA - QUANTITATIVE PARAPLEGIA DATA ...................................................................... 49 EPILEPSY................................................................................................................................... 51 CAMEROON – QUANTITATIVE EPILEPSY DATA........................................................................... 51 AUSTRALIA - QUANTITATIVE EPILEPSY DATA ........................................................................... 52 COMPARING THE COUNTRIES .............................................................................................. 54 PARAPLEGIA – COUNTRY COMPARISONS ................................................................................... 54 EPILEPSY – COUNTRY COMPARISONS ........................................................................................ 55 COUNTRY BY DISEASE .............................................................................................................. 55 CHAPTER 4 DISCUSSION....................................................................................................... 58 LIMITATIONS OF THE STUDY DESIGN ......................................................................................... 58 Comparing “like” health conditions ................................................................................... 58 Sampling.............................................................................................................................. 59 Cross cultural validity ......................................................................................................... 60 Self-report data ................................................................................................................... 60 IMPLICATIONS ........................................................................................................................... 61 Health is context independent – or no-context is the right context...................................... 61 Context and egalitarianism are incompatible. .................................................................... 64 CONCLUSION............................................................................................................................. 65 CHAPTER 5 A RESEARCH AGENDA................................................................................... 68 REFERENCES............................................................................................................................ 71

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ACKNOWLEDGEMENTS The authors would like to offer their sincere gratitude to the participants and their families for their time, effort and contribution to this study. We would also like to thank the board, programme officers and staff of the Epilepsy Foundation of Victoria, particularly Margot Boyle and Russell Pollard; the board and administrative staff of Wheel Chair Sports Australia, particularly Mr Darren Cunningham and Mr Phil Tracey, and the staff of the ParaQuad Association, particularly Janice Florence. Joseph Toscano and Margaret Kiriakidis provided crucial assistance in accessing participants from the community. The Cameroon team are very grateful to all who facilitated the study including Dr Akoto Eliwo, Director of IFORD and Mr Eloundou Enama, Director of National Centre for Rehabilitation of the Handicap Yaounde (CNRH) who made it possible for the research team to get in contact with former patients of his institution. Sr Mary Vejai, Director of Saint Joseph children and Adult Home (SAJOCAH) Bafut and Yuh Simon, Director of Community Based Rehabilitation Centre Mbingo facilitated access to rural paraplegics. Mr Siandé Robert also helped the research team in the identification of rural paraplegics in some villages in the Central province. Mr Kabamba Emmanuel, assisted the research team with access to the urban and rural health centres where epileptics were contacted and given appointment for interviews. He also facilitated access to the various villages in the rural area. This research was carried out with the invaluable assistance of Kerry Hollier in Melbourne and Dr Innocent Takougang, Mr Tatah Peter Ntaimah, Miss Tumi Patience Njong, and Dr Martyn Sama in Yaoundé. Joni Law and Siang Tia from the Key Centre for Women's Health in Society provided crucial administrative support and Lenore Manderson, Director of the KCWH, gave invaluable intellectual feedback and editorial comments. This research was funded by grants from the Global Forum for Health Research and the University of Melbourne through the Melbourne Research Career Establishment Grant Scheme.

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EXECUTIVE SUMMARY The Disability Adjusted Life Year (DALY) is a measure of population health that was developed, as part of a World Bank initiated study, to inform health strategy development, priority setting, resource allocation and research, and to measure the global burden of disease. The innovative feature of the DALY was the combination of information on morbidity and mortality within a single index. Since its development in 1992, the rate at which it has been adopted by governments, multilateral agencies and researchers has been staggering. The enthusiasm with which the measure has been taken-up perhaps reflects a desire on the part of health policy makers to embrace an “evidence-based” approach to health policy. Although the DALY has been heavily criticised in some quarters, it has survived. There are a number of reasons why this may be: •

It is a good measure of population health;

•

The enormous political will to see it succeed; and

•

The lack of empirical data to challenge the validity of the DALY;

•

In addition, many of the criticisms of the DALY have been about the implicit and explicit social values, which are hard to argue on technical grounds.

This report details the findings of an empirical investigation of some of the technical and social assumptions on which the DALY is based. The specific aim was to examine the justification behind the use of a single global severity weight for each health condition without regard to contextual factors. The objectives of the study were to examine the notion that the burden of a disease is broadly similar without regard to country, environment, gender or socioeconomic status, and to develop detailed descriptions of the experiences of the burden of disease as they related to these contextual factors. Serious technical deficiencies with the global severity weight are raised in Chapter 1.* We argue that the global severity weight will incorrectly estimate the burden of disease, unless the local average social milieu in which a health condition occurs is the same as the global average social milieu. That is to say, if the average handicap within a local context is different from the average handicap in a global context, the DALY cannot be used in a global burden of disease exercise and may seriously misinform policy makers. In Chapter 2 we describe the design and protocols for a multi-factorial, exploratory study employing qualitative and quantitative techniques, which was used to examine the evidence for DALY. The effects of country (Cameroon or Australia) by environment (urban or rural) by gender, and by socioeconomic status on two health conditions (epilepsy and paraplegia) were examined. A total of 160 participants were recruited for the study and data were collected using a protocol combining instruments for measuring subjective quality of life (ComQoL) and health-related quality of life (SF12), with qualitative techniques such as Photo Voice and in-depth interviews. The data

*

It should be noted that many of the criticisms made of the DALY in this report could also be made of the Disability Adjusted Life Expectancy (DALE), because both measures rely on the same approach to the estimation of the severity weight for each health condition.

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provided an extensive and detailed compilation of context-rich descriptions of living with epilepsy or paraplegia. The striking features of the data were the enormous differences between countries with respect to the impact of the health conditions on functioning. These results are described in Chapter 3. The lack of infrastructure in Cameroon in general and in the rural areas in particular made coping with paraplegia extremely difficult. People with paraplegia commonly die of generalised infections arising from pressure sores, within two years of their initial injury; indeed, in Cameroon paraplegia is generally regarded as a terminal condition. Detailed descriptions were provided by participants of crawling through human waste in order to access pit latrines, and of being confined to a single room that served as a bedroom, living room, toilet and kitchen. The stench produced by these single room existences resulted in social isolation, depression and other mental health problems. Suicidal ideation was expressed in a number of cases. Given that the people interviewed had been paraplegic for at least five years, the suicidal ideation was of concern and contrary to the expected adaptation and coping that frequently occurs with chronic conditions. People of higher socioeconomic status had some advantages because of their ability to purchase the goods and services necessary for personal care and to improve their level of functioning. Both the qualitative and quantitative tools revealed that people with paraplegia in Australia were substantially better off than people in Cameroon. Due to the nature of sampling, several participants in the study were elite athletes and data collection coincided with a strong national celebration of disabled sportsmen and women leading to the Paralympic 2000 in Sydney. However, there was substantial variation in the experiences of the paralympians and other people with paraplegia who were interviewed. Experiences ranged from poor treatment within the health and social welfare services to discrimination from the general population. Pain was a common complaint, which they reported tended to be largely ignored because of the assumption that there was no feeling with paralysis. There was also a fragmentation of medical care, which led to individual symptoms being treated without regard to the whole person, and one of the health-related problems that was least well managed was incontinence. Within the group there was a marked difference in the quality of and access to services in rural communities. It was also evident that participants who were financially well off, could buy the equipment and services they required to improve their quality of life. People with epilepsy in Cameroon suffered high levels of discrimination because of the cultural and social stigma associated with the condition. This affected all aspects of their life and consequently their health. They were unable to generate income and they had little physical or emotional support from family and community. The lack of availability, cost and the difficulties of access to services further limited their ability to improve their health status. Socioeconomic status, gender and environment did not make a significant difference on the quantitative measures. However, the differences apparent in the qualitative data suggested that if people who knew the condition but were unfamiliar with these contextual factors were asked to evaluate the severity of the condition, they would evaluate the severity differently for different contexts. For women, single parenthood was common because marriage to people with epilepsy was not acceptable socially or culturally. Financial constraints made obtaining health care and regular medication, as well as coping with the side effects of anti-epileptic drugs, a major problem. A relatively common co-morbidity reported was severe burns as a result of falling into open fires (used for cooking) during seizures.

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People with epilepsy in Australia scored lower than the population norms on the subjective quality of life measures. Qualitative data demonstrated that a major issue, in addition to some of the discrimination and stigmatisation issues articulated by participants from Cameroon (although to a lesser degree), was coping with the side effects of the anti-epileptic medications. Commonly reported side effects were headaches, drowsiness, dizziness, and forgetfulness. The side effects were severe enough for participants to contemplate the relative advantage of occasional seizures and less medication. A comparison of the subjective quality of life scores for Australians with epilepsy or paraplegia indicated that people with paraplegia were “better off” at least with respect to subjective quality of life. This was in direct contrast to the severity weights used in the Australian burden of disease study that ranks the severity of paraplegia as worse than that of epilepsy. The situation was reversed in Cameroon, however, where the subjective quality of life for people with paraplegia was worse than for those with epilepsy. This apparent reversal of the rank order of the severity of two health conditions between countries again suggests the possible failure of the global severity weight as a coherent concept. The meaning of these data in the context of the DALY is discussed in Chapter 4. The data collected were not without their problems, and these could explain some of the observed differences. It was apparent, however, that where interpretive difficulties arose in the quantitative data because of methodological problems, the qualitative data were of very high quality and could usually resolve these difficulties. There is little doubt about the broad thrust of the findings. The justification for the uncritical application of a global severity weight in the measurement of population health is absent or very weak; and the use of the global severity weight is likely to reinforce already existing inequalities in health. The defence that a single weight for each health condition is needed for the comparison of countries, sexes, or age groups is manifestly untrue. Given that the level of development within countries appears to have a significant effect on the burden of disease, the current global estimates of the burden of disease disaggregated by country almost certainly overestimates the burden within developed countries and underestimates the burden in developing countries. Consideration is given to a range of other defences, including the notion that: •

“Contextualised” severity weights defeat the “egalitarian” principles on which the DALY is based;

•

Contextualised weights prevent comparison between regions;

•

Contextualised weights are too expensive to derive;

•

An alternative definition of “health” that avoids “social milieu” resolves the problem; and

•

The use of separate weights for treated and untreated individuals resolves the problem.

A research program that could provide the evidence to overcome some of the technical deficiencies of the DALY is proposed in Chapter 5. This program starts from the premise that the burden of disease can only be measured accurately if the contextual

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information that creates the burden associated with a disease becomes one of many foci of health research. Unfortunately, there is very little data on how social, cultural and environmental factors affect the severity of a health condition. More importantly, there is a paucity of information about how this data can be rapidly and validly collected so that it can be fed into the process of estimating the global burden of disease. There have been some attempts recently to develop culturally appropriate techniques for obtaining population estimates of the severity of health conditions in developing countries. These start with a preconception of how health and disease must be viewed in order to obtain appropriate measures. However, this research does not address what would seem to be a fundamental issue: very little is actually known about the impact of disease in different social, cultural, and environmental settings. Some of the stigmatisation research has begun to gather these data, but the focus has been narrow. If the global burden of disease is to be measured, and resources to be appropriately allocated, a broad research program is needed to document the effect of context on the severity of disease. To do this, the research program would need to expand the number of conditions under consideration, the number of countries under consideration, and increase the sample size for each health condition. Questions to be answered would include: • •

•

What is the best method for obtaining valid descriptions of health states within contexts? How can one render contextualised descriptions of a health condition for severity weight estimation? How should one estimate the unique and shared contribution of the factors to the severity weight?

G The World Bank has shown leadership in its attempt to

develop better methods for measuring the Global Burden of Disease.

G The DALY has serious technical deficiencies that result in

the estimates of the burden of disease being wrong.

G The deficiencies in the DALY are likely to exacerbate

health inequalities, such that the burden of disease in developing countries is underestimated, and the burden of disease in developed countries is overestimated.

G For the study of the global burden of disease to be

meaningful, the effect of social, cultural, and environmental factors on the impact of disease will increasingly have to become the focus of research.

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RÉSUMÉ Le DALY (Disability Adjusted Life Year) est une mesure de l’état de santé de la population développée récemment. Son originalité réside dans la combinaison des mesures traditionnelles de mortalité et de morbidité. Parce qu’il permet de mesurer le fardeau de la maladie, cet indicateur sert à différentes fins comme l’orientation de la stratégie de santé, la définition des priorités de recherche et d’actions, l’allocation des ressources, etc. Depuis sa formulation en 1992, il a servi de base aux décisions de nombreux gouvernements, des agences de développement ainsi qu’aux travaux de nombreux chercheurs. L’enthousiasme avec lequel il a été utilisé au cours des dernières années traduit la volonté des décideurs politiques de baser leurs actions sur la réalité des faits. Cependant, l’indicateur porte en lui de nombreuses limites décriées par de nombreux auteurs. Ces nombreuses critiques n’ont, pourtant, jamais pu atténuer cet enthousiasme. Plusieurs raisons peuvent expliquer cela: • • • •

L’excellence de la mesure La forte volonté politique qui a sous-tendu son développement La faiblesse technique de certaines critiques qui ne traitent que des valeurs sociales Les critiques formulées sur la validité de l’indicateur reposent rarement sur des données empiriques.

Ce rapport de recherche présente les résultats d’une étude empirique de certaines des hypothèses et de certains aspects techniques de la construction du DALY. L’objectif de l’étude est de tester l’hypothèse selon laquelle le fardeau de la maladie est invariable quels que soient le pays, l’environnement, le genre, ou le statut socio-économique. Elle vise aussi à décrire concrètement le fardeau de la maladie tel qu’il peut-être associé à ces différents facteurs contextuels. L’objectif spécifique est de tester la vraisemblance de cette hypothèse en examinant le bien fondé des raisons qui sous-tendent le choix d’un taux unique de gravité pour chaque maladie. Dans le chapitre 1 sont traitées les déficiences techniques de cette hypothèse. Il est, en effet postulé, que , à moins que les conditions moyennes du milieu sociale soient identiques aux conditions globales de ce même milieu, recourir à un taux global de gravité conduit à une estimation erronée du fardeau de la maladie. Plus spécifiquement, si l’invalidité moyenne d’un contexte local donné diffère de celle prévalant dans un contexte global, alors le DALY ne peut être utilisé pour mesurer le fardeau global de la maladie. En y recourant, on court le risque d’induire les décideurs en erreur. Le chapitre 2 décrit la démarche méthodologique de l’étude. Elle consiste en une étude exploratoire recourant à la fois aux méthodes quantitatives et qualitatives. Les données collectées permettent de saisir l’effet du contexte du pays (Cameroun et Australie) sur le fardeau de la maladie, selon le milieu de résidence, le genre, et le statut socio-économique. Au total 160 personnes ont été interrogées à partir d’un questionnaire comportant différents volets permettant de mesurer la qualité de la vie (subjective) et les conditions (ou l’état) de santé. La qualité de la vie est mesurée par un outil couramment utilisé par les psychologues: le «Comprehensive quality of life» et l’état de santé par un outil couramment utilisé par les spécialistes de santé: le SF12. Le questionnaire comporte aussi des outils qualitatifs comme le «photo voice» et un guide d’entretien approfondi. Les données collectées offrent un large éventail de situations dans lesquelles vivent les épileptiques et les paraplégiques. Il ressort de ces données une différence notable entre pays en ce qui concerne l’impact de la maladie sur la capacité de fonctionnement des personnes enquêtées.

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Le chapitre 3 décrit ces résultats de manière plus détaillée. L’absence d’infrastructure adaptée à la condition des paraplégiques rend leur vie quotidienne très difficile, plus particulièrement en milieu rural. Les paraplégiques meurent souvent d’infections généralisées, consécutives aux escarres, au bout de deux ans après leur accident. Au Cameroun cela est généralement considéré comme l’étape finale. Les participants vivent dans des conditions d’hygiène lamentables avec des latrines entourées d’ordures. Ils sont souvent confinés dans une pièce unique servant à la fois de chambre, de salon, de toilette et de cuisine. Les odeurs nauséabondes que dégagent ces pièces entraîne l’isolement des malades qui les conduit à la dépression et à des problèmes de santé mental aigus. Il arrive souvent que certains pensent au suicide. Parce que les paraplégiques interviewés le sont devenus depuis au moins 5 ans, cette tendance au suicide est préoccupante. En effet, il a été observé qu’en général, les malades chroniques s’adaptent mieux et font mieux face à leurs conditions à mesure que passe le temps. On constate que ce n’est pas le cas des paraplégiques Camerounais. Toutefois, les paraplégiques ayant un statut socio-économique élevé sont avantagés de ce point de vue parce qu’ils disposent des ressources pour accéder aux services et se procurer le matériel dont ils ont besoin pour assurer leur fonctionnement. Aussi bien les données quantitatives que les données qualitatives révèlent que les paraplégiques australiens sont mieux lotis que ceux du Cameroun. La solution aux difficultés de terrain rencontrées a conduit à sélectionner des paraplégiques parmi des athlètes d’élite devant participer aux jeux olympiques pour handicapés de Sydney 2000. Toutefois, les para-olympiens semblent partager les mêmes expériences que les autres paraplégiques australiens. Ces expériences vont du mauvais traitement dans les services de santé et sociaux et à la discrimination dont ils sont l’objet dans la population en général. Ils se plaignent souvent de douleurs intenses qui sont ignorées par les proches parce qu’on suppose que leur paralysie les empêche de sentir la douleur. Ils se plaignent aussi de la fragmentation des soins médicaux qui fait que les symptômes individuels sont traités sans beaucoup de considération pour la personne humaine. Par ailleurs, le problème de l’incontinence chez les paraplégiques ne semblent pas traité avec tout le sérieux nécessaire. Dans le groupe, il y a une forte disparité dans l’accès aux services en milieu rural. Il ressort aussi des données que les plus nantis peuvent se procurer le matériel nécessaire à l’amélioration de leur qualité de vie. Les épileptiques du Cameroun souffrent énormément de discrimination à cause du stigma culturel et social lié à la maladie. Cela affecte les différents aspects de leur vie et en conséquence leur santé. Ils n’ont pas d’activité génératrice de revenu et reçoivent très peu de soutien physique et moral de leur famille et de la communauté. L’absence de service, les coûts élevés de ces services lorsqu’il y en a et leur accès difficile limitent la capacité des épileptiques Camerounais à améliorer leur état de santé. Les données quantitatives ne permettent pas d’établir une différence significative entre les groupes constitués selon le statut socio-économique, le sexe et le milieu de résidence. En fait ces différences existent, mais elles ne sont pas statistiquement significatives. Toutefois, l’analyse des données qualitatives leur donne un sens. Elles révèlent en effet que la gravité de la condition des épileptiques varie d’un groupe à l’autre. Parmi les femmes, on rencontre beaucoup de monoparentales parce que le mariage avec une épileptique est culturellement et socialement non toléré. Les contraintes financières rendent l’accès aux soins, l’acquisition des médicaments, et la lutte contre les effets secondaires problématique. Les épileptiques sont souvent exposés aux brûlures dues à la chute dans le feu lors de la préparation des repas. Le niveau de qualité de vie des épileptiques Australiens est inférieur à celui de la population normale. Les données qualitatives révèlent qu’en plus des problèmes observés chez les épileptiques Camerounais, ceux de l’Australie ont des difficultés à

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faire face aux effets secondaires des médicaments anti-épileptiques. Les effets secondaires les plus mentionnés sont: les maux de tête, la somnolence, l’étourdissement, et les pertes de mémoire. Ces effets secondaires sont parfois si forts que certains malades leur préfèrent les chutes occasionnelles que provoque la non prise d’anti-épileptiques. Il est intéressant de constater que les paraplégiques Australiens ont une qualité de vie supérieure à celle des épileptiques. Ceci est en contradiction avec le taux de gravité attribuée à ces deux maladies, ce dernier étant plus élevé pour la paraplégie. Au Cameroun, on constate l’inverse, avec une qualité de vie inférieure chez les paraplégiques. Ces résultats contradictoires entre deux pays pourraient traduire l’impertinence d’attribuer un taux de gravité unique quel que soit l’environnement. On en déduit l’incohérence du concept de taux global de gravité. Le chapitre 4 discute les résultats présentés ci-dessus. Les données collectées peuvent certes avoir quelques limites, et celles-ci ont pu entacher certains des résultats. Les difficultés d’interprétation des données quantitatives qui pourraient en résulter sont toutefois facilement résolues par les données qualitatives qui elles sont d’une très grande qualité. Il ne peut donc y avoir de doute sur la fiabilité de nos résultats. Ces résultats ne supportent pas l’hypothèse d’un taux unique de gravité associé à une maladie. Il eût été plus indiqué d’envisager différents taux correspondant aux différents milieux sociaux. Le recours à un taux unique pour le calcul du fardeau associé à une maladie quelconque pourrait renforcer les inégalités déjà existantes dans le domaine de la santé. L’hypothèse selon laquelle un seul taux de gravité est nécessaire pour comparer les pays, ou différentes catégories sociales à l’intérieur d’un même pays est invraisemblable. Etant donné que, selon nos résultats, le niveau de développement du pays affecte de manière significative le fardeau de la maladie, les estimations actuelles qui en sont faites et qui sont par la suite désagrégées par pays, surestiment le fardeau de la maladie dans les pays développées tout en le sous-estimant dans les pays en développement. D’autres considérations relatives au calcul du DALY ressortent de nos résultats: •

Les taux de gravité calculés en tenant compte du contexte l’emportent sur ceux reposant sur le principe d’égalité comme c’est le cas du DALY;

•

Les taux contextualisés rendent inutile la comparaison des groupes;

•

Les taux conceptualisés sont coûteux à obtenir;

•

Cela pourrait expliquer pourquoi on recourt à une mesure qui fait abstraction du contexte dans la définition de la santé; et

•

Le recours à des taux distincts pour les personnes traitées et celles qui ne le sont pas résout le problème.

Un programme de recherche offrant les informations permettant de corriger les déficiences techniques du DALY est proposé au chapitre 5. Ce programme repose sur la prémisse que le fardeau de la maladie ne peut être mesuré correctement que si les caractéristiques du contexte qui contribuent à la création de ce fardeau font partie des éléments sur lesquels l’accent doit être mis dans la recherche sur la santé. Malheureusement, l’on dispose de très peu d’information sur la manière dont les facteurs sociaux, culturels et environnementaux affectent la gravité de la maladie. Plus encore, on ne sait pas encore très bien comment collecter rapidement de telles données, de manière à ce qu’elles servent directement au calcul du fardeau de la maladie.

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Des tentatives ont été faites récemment pour développer des méthodes permettant de mesurer la gravité de la maladie qui tiennent compte du contexte culturel dans des pays en développement. Elles partent du présupposé que la manière dont la santé et la maladie sont perçues compte dans une bonne mesure de la gravité de la maladie. Malgré son intérêt certain, cette recherche occulte l’aspect le plus important consistant à savoir quel est l’impact de la maladie dans différents contextes sociaux, culturels et environnementaux. Certaines recherches sur les stigmas commencent à offrir de telles informations, mais leur portée semble limitée. Pour que le fardeau de la maladie soit correctement mesuré et qu’il puisse servir de base à l’allocation des ressources affectées à la santé, un programme global visant à déterminer l’impact du contexte sur la gravité de la maladie est nécessaire. Une telle recherche doit porter sur un grand nombre de maladies, concerner plusieurs pays, avec des tailles d’échantillons suffisamment élevées. Ce programme permettra de répondre, en autres, aux questions suivantes: • •

Quelle est la meilleure méthode pour obtenir une bonne description de l’état de santé dans différents contextes? Comment peut-on se servir de la description contextualisée d’une condition de santé (maladie) pour estimer le taux de gravité? et

Comment peut-on estimer la contribution de la part unique et la part commune des différents facteurs au taux de gravité?

G

G

G

G

La Banque mondiale a joué un leadership dans la quête de meilleures méthodes pour la mesure du fardeau global de la maladie. Le DALY porte en lui de sérieuses déficiences techniques qui conduisent à une estimation erronée du fardeau de la maladie. Ces déficiences pourraient aggraver les inégalités déjà existantes, en ce sens qu’elles aboutissent à une sousestimation du fardeau de la maladie dans les pays en développement et à sa surestimation dans les pays développés. Pour être pertinente toute étude sur le fardeau global de la maladie doit mettre l’accent sur l’effet des facteurs sociaux, culturels, et environnementaux sur l’impact de la maladie.

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CHAPTER 1 INTRODUCTION A good measure of population health is critical to the practice of public health. Measures of population health can inform the development of health strategies and health priorities, resource allocation, and research. The “Disability Adjusted Life Year” (DALY) is one such measure, developed as a means of quantifying the burden of disease and injury at the level of populations [1]. This relatively new metric was promulgated by the WHO and the World Bank as a means of combining information about mortality and morbidity within a single index [2]1. The growing importance of the DALY can be seen in its use in such areas as the development of “evidence-based health policy” [5], the general estimation of the global burden of disease [6], the identification of emerging health problems [7], the determination of research priorities [8, 9], and to guide investment policies for health [10]. Increasingly, it is being adopted by national and state governments to assist in an “evidence-based” approach to health resource allocation (e.g., the Chilean government [11] and the Australian Government [10], and at the state level, the Victoria State Government [12]). There are also a growing number of studies, not directly sponsored by governments, which use the DALY methodology to estimate the burden of disease [13-15]. Some have suggested that the DALY is so robust that it will become something of a de facto standard for the official reporting of health outcomes [16]. This report summarises an empirical investigation of the validity of some technical and some arbitrary (or what Bobadilla [17] described as “social preference”) aspects of the DALY. This chapter starts with a discussion of measures of population health. It then focuses on the limitations of measures based on mortality and leads into a description of the development of the DALY. A concise summary of some criticisms of the DALY are then presented – neither doing justice to the criticisms, nor the counterarguments. This is done to provide a flavour of the current debates on the topic. The focus is then narrowed to one aspect of the DALY, namely the global severity weight.2 The narrowing of focus is initiated with a general discussion of the use of the global severity weight. This is followed by a critique or “deconstruction” of the concept. Finally, the general questions to be answered by the current study are discussed.

Measures of Population Health The DALY is not the only measure of population health. One proxy measure of population health common to the late 20th century is the mortality rate. One needs to look no further than recent epidemiological texts (e.g., [19, 20]) or compilations of indices ([21]) to see that mortality remains a widely used indicator of population health. This should come as no surprise because of the simplicity of this measure. Death does not (generally) require diagnostic expertise; there are no subtle variations of death that one needs to explicate; the dead are simpler to count than the wounded; and mortality statistics are easier and more likely to be routinely maintained by the state than are morbidity statistics. One particular mortality-based measure of population health that has, in the past, been regarded as a highly sensitive measure is infant mortality [22, 23]. “…[I]nfant mortality has always been acknowledged to be a sensitive indicator of a group’s or a society’s general health status. Because a decline in infant mortality has, in

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the past, been one of the most marked features of a nation’s economic development, the rate has been looked at as one of the most important measures by which the living conditions or social well-being of countries, or groups within countries, could be compared…. Generally it is agreed that high infant mortality is an indicator of poor environments, high rates of illness and possibly poor conditions for birth.” [22] (p.34)

Proxy measures of population health such as infant mortality are not, however, without their problems. Frequently starting as indicators of a whole population’s general health, they often become the principle focus of health policy such that health strategies and health priorities are reformulated with the (proxy) outcome measure in mind [2]. Health policies begin explicitly to target the chosen outcome measure, while ignoring the rest of the population, for which the outcome measure was supposed to be an indicator. Thus, infant mortality may drop, because it becomes the principle focus of health policy, but the population’s health may remain static or even degrade [2]. Paradoxically, if a measure [of population health] is used, it will influence policy debate, permeate the thinking of decision makers and become part of the culture of the subject. In other words, an indicator that is widely used will soon become normative through its use [2] (p.3).

There are other problems associated with mortality as an indicator of population health. For instance, it undervalues health outcomes that are severely disabling, but do not result in death. As medical interventions improve, so one can avert death, but not necessarily morbidity, and this results in an overestimation of a population’s health. In effect, survival rates rise, but the quality of life of the survivors may be deplorable [24].

Developing the DALY Health policy makers are therefore left in something of a quandary. Mortality is not a good measure of population health because it ignores morbidity. Proxy-measures for a population’s health tend to focus policy on the minimisation of the poor health outcomes reflected by that measure to the possible exclusion of other poor health outcomes. What is required is an objective and “true” (i.e., not a proxy) measure of a whole population’s health that accounts for both the mortality and morbidity attributable to disease. The DALY was motivated by the perceived need: •

To develop a single measure of the burden associated with a disease that combined years of life lost due to premature mortality and years of life lived with disability (or healthy years lost due to disability) [3]; and

•

To decouple epidemiology from advocacy, thereby creating an objective measure of the mortality and morbidity attributable to different diseases [2, 25].3

The DALY is a time-based measure accounting for years of life lost due to premature mortality and healthy years of life lost due to disability [6]. The years of life lost for a given health state, i, are calculated as: DALYi = YLLi + YLDi, where YLLi is the years of life lost in a population due to premature mortality attributable to health condition i, and YLDi is the healthy years of life lost in a population due to disability attributable to health condition i. The measure is moderated by four factors [25], as follows:

2

•

There is a 3% annual discount rate attached to the years of disability or premature mortality.

•

There is an age weight such that deaths or disabilities occurring in childhood or old age accrue fewer DALYs than deaths or disabilities occurring in the middle years of life. This is to take account of the perceived greater social value of people in their middle years of life.

•

There is a sex weight such that two people of the same age with the same disabling condition but of different sexes accrue a different number of DALYs. This is to account for the biological differences in life expectancy of males and females, such that for the same condition, females with a particular health condition accrue more DALYs than males. The distinction needs to be made between a social preference based on gender and a biological difference based on sex. It is the latter that is included in the DALY.

•

Each health condition has an associated severity weight in the interval (0, 1). A health condition with a severity weight of 0 has no adverse impact and a condition with a severity weight of 1 is equivalent to death. The adverse effects of a health state are judged, in part, according to the definition of disability provided in the International Classification of Impairments, Disabilities, and Handicaps (ICIDH) [26].

•

There are several papers on the DALY that review its methods and purpose and readers may refer to those for more specific technical clarification [2, 3, 6, 25, 27, 28].

Criticisms of the DALY Notwithstanding the growing influence of the DALY in population health, it has not been without its critics [17, 27-41]. Some have criticised it on grounds of equity [27, 28, 32, 38], others for its failure to account adequately for the burdens associated with specific diseases or in specific populations [29, 30, 36, 37]. Arnesen and Nord [31] and Groce et al. [39], criticised it because it explicitly values the life of a disabled person as less than that of a person without a disability. AbouZahr has criticised the measure for its failure to account for the burden associated with late foetal deaths [29].4 Some critics seem to regard the problems with the DALY as so devastating as to make the measure unusable [27, 28, 37, 41]; others see the problems as opportunities for corrective action [29, 30]. In a brief summary, Bobadilla divided the criticisms that the DALY has attracted in two broad areas [17] (p.7). There are criticisms that relate to technical deficiencies and criticisms that relate to the social preferences on which the DALY is based. Social preferences, such as the age weighting and the 3% discount rate, cannot be justified on technical grounds [17].

The Global Severity Weights (GSW) One aspect of the DALY that does not seem to have attracted as much critical comment as other aspects is the use of the global severity weight. AbouZahr [29], and Barker and Green [32] criticised the severity weight because of the the focus on “disability” rather than “handicap” in the construction of the severity weight. Bobadilla [17] noted that:

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The disability weights [global severity weight] ignore the handicap attached to some permanent disabilities in different societies. The same disability has different effects on the lives of individuals in different countries. Some traditional societies stigmatise and reject individuals with specific permanent impairments, such as infertility, AIDS or psychosis. The real health loss in these individuals is greater than that estimated by the DALY…[T]his poses a problem for national burden of disease assessments. (p.7)

A much more pertinent argument, and one we take up in more detail shortly was raised by Sayers and Fliedner [41]. They criticised the failure to take account of regional variation in the distribution of social circumstance that contribute to handicap. “Disparities across communities arising from these factors may be wide spread and result in complex population-wide heterogeneity” (p.383)5. The use of a single global severity weight for each health condition has been argued by the authors of the DALY on three grounds. The first argument is that a single global severity weight for each health condition is the basis for maintaining an objective measure that is comparable between regions [25, 42, 43]. It is this argument that is addressed in the following section. Second, the use of a global severity weight is computationally convenient [25]. It seems self-evident that if one adopts simplifying assumptions, it will make things easier. We consider the costs and benefits of this in the final chapter. The third argument, in support of the global severity weight, is that it supports the principles of equity that underpin the DALY [2, 3, 42]. In Bobadilla’s terms, the first two arguments are technical and the third relates to social preference. We defer considerations of the latter argument until the final chapter, when theory and data are brought together.

Deconstructing the GSW In this section, we consider the technical support for the proposition that one needs a single global severity weight for each health condition in order to make comparisons between countries on the burden of disease attributable to that condition. Before examining the notion of a global severity weight in any detail, however, we wish to consider first the purpose of estimating the global burden of disease for any, or all, diseases. Knowing that the global burden of disease attributable to AIDS is x, or that AIDS accounts for z% of the total global burden of disease in and of itself, is of only limited use. For the estimate to be more useful one needs to be able to disaggregate it (e.g., by region), in much the same way as was done in The World Health Report 2000 [44]. The disaggregated data provides information about where the need lies for intervention. There would, for instance, seem to be little point intervening with equal effort globally on AIDS when the majority of the cases are occurring in Africa.

Background to the GSW The purpose of a global severity weight for each health condition is to reflect the impact of having that health condition. Each weight was estimated using an iterative process with a panel of judges6. The idea behind the severity weights was to restrict the consideration about the severity of a condition to the definition of disability according to the International Classification of Impairments, Disabilities, and Handicaps’ (ICIDH) [26]. In that document, “disability” was defined thus:

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In the context of health experience a disability is any restriction or lacking (resulting from impairment) of ability to perform an activity in the manner or within the range considered normal for a human being. [26] (p.28)

However, in the weight estimation exercises, judges were also asked to consider the “average conditions of individuals and social responses” or “average handicap” ([2], p.34) where, again, the ICIDH definitions were used. “Handicap” was defined thus: In the context of health experience, a handicap is a disadvantage for a given individual, resulting from an impairment or disability, that limits or prevents the fulfilment of a role that is normal (depending on age, sex and social and cultural factors) for that individual. [26] (p.29)

For example, when estimating the severity weight for deafness, the panel was to have taken account of both the functional loss associated with deafness (i.e., the disability) and the impact that the “average conditions of individuals and social responses” [2] (p.8) or “average social milieu” would have on deafness.7

The “average social milieu” There has, unfortunately, been no substantial discussion of what it means to take account of the “average social milieu”. At its most meaningful, it is either (a) the average social condition of the population as a whole or (b) the average social conditions in which people with a particular health condition live.8 These two constructions are distinct, and the distinction needs to be understood. Furthermore, for “social milieu” to be useful for a realistic assessment of the burden of disease, it would also need to encompass a broad range of cultural, economic, gender, and environmental factors. A broad reading of “average social milieu” would certainly be in keeping with the notion of “average handicap”. In discussing these points over the next few paragraphs, sentences are marked ‘a’ or ‘b’ to indicate whether the point relates to the average social condition of the population (a) or of the people with the particular health condition (b). Imagine that the curve in Figure 1 below (ignoring the shading) shows the distribution of “social responses” in a particular population (a). In this population, social response tends to be more negative (the left tail) than positive (the right tail). The colored shading shows the distribution of some health condition, x, across the population. Most people with health condition x live among people with a negative social response (i.e., the darker shading in the curve), and very few people with health condition x live among those with a positive social response (i.e., where the curve is shaded white) (b). The problem with using the average social milieu of an entire population is that the average person who has the health condition may not be the average person in the population. They may, thus, not be exposed to the average social conditions in the population as a whole, but to some more extreme version of the population’s social response. Given that it is unlikely that health conditions are distributed over an entire population in the same manner in which social responses are distributed, if the panel takes account of the average social milieu of the entire population (a), when making a judgement about a health condition, the estimate of the severity weight would be inaccurate9. In order to estimate correctly the severity weight for each health condition, the panel would need to take account of the average social milieu applicable to people living with that particular health condition (b). However, the average social milieu would be very different for different health conditions. For example, heart disease is more common in the elderly and the social milieu within which older people exist is

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likely to be very different from the social milieu of the much younger population of people with infertility, for instance. For each health condition’s severity weights to be accurate, a panel would need to understand what the average social milieu was for each health condition. This is an important technical problem for the DALY.

Figure 1 The distribution of a health condition x (the shading) overlaying the distribution of social responses in a population

Making global comparisons using a GSW Let us assume, for argument’s sake, that one can ground the panel judges in the (global) average social milieu of each health condition and thereby obtain a good (i.e., accurate) assessment of the global severity weight for a particular health condition. A second technical problem arises. This second problem relates to the futility of using a parameter estimated on the basis of aggregated data to provide any information about the magnitude of the parameter for some disaggregated unit of the data. That is, a parameter estimated on the basis of the whole world does not tell you anything about the magnitude of the parameter in any one region. Yet one of the reasons for using the DALY in the global burden of disease study was to inform national policies. The estimation of the burden of disease for a disabling condition is the sum of all healthy years of life lost (globally) due to disability (appropriately adjusted for sex, age and time) multiplied by the severity weight. The severity weight that is based on the global average social milieu of people with the health condition need not be true for any single region, sub-region, or country. If one tries to compare the burden of disease for regions or countries using a single global severity weight (e.g., [45]), the implication is that the severity weight applicable in each disaggregated unit is the same. For example, if one is comparing the burden of disease attributable to deafness between countries, then the average social milieu for people living with deafness in Sweden must be same as the average social milieu for people living with deafness in Burkina Faso. By way of elaboration, consider the following example of a hypothetical disease, xitis, which is disabling but not fatal. If xitis occurs in only one country, then the appropriate global severity weight for xitis is the same weight as the appropriate local severity weight. If xitis occurs in two countries (Atlantis and Nirvana), then the

6

appropriate global severity weight is either (a) a synthetic estimate based on a panel judgement, such as that used in the Global Burden of Disease study, or (b) it is a weighted average of the separately derived synthetic estimates of the severity weight in Atlantis and the severity weight in Nirvana. If by chance, the Nirvana severity weight and the Atlantis severity weight are the same, then they will obviously equal the global severity weight and disaggregating burden of disease estimates will be unproblematic. If however, the severity weights are not identical, then the application of the global severity weight will overestimate the burden of xitis in one country and underestimate it in the other. Notwithstanding the argument presented above, the idea of comparing the burden of a disease between countries when the estimates were based on different severity weights has been criticised [2]. Murray and Acharya wrote on this point, “If the burden estimated from a given condition were to be completely context specific, meaningful comparisons across communities or within communities over time would be essentially impossible” [25] (p.726). Another DALY author recently reiterated the argument (A. Lopez, Personal Communication, October 2000, Bangkok). He suggested that local governments may choose to use locally derived severity weights for local burden of disease studies, but for the purposes of international comparison, a single severity weight for all countries had to be used. To do otherwise, he argued, would result in a comparison of “apples with oranges”. The arithmetic, however, defies the argument and raises important questions about the validity of the burden of disease estimates based on a global severity weight. In the World Health Report 2000 the DALYs attributable to different health conditions are given globally and disaggregated by region [44] (p.170). All other things being equal, the global estimate of DALYs could be correct only if the synthetic panel estimate of the severity weight for each condition were correct. The DALY estimates for each region, however, would only be correct if the regional severity weight for each health condition were equal to the global severity weight. This would generally only be the case if the average social milieu within each region were the same as the global average social milieu. We leave the discussion of the global severity weights at this point, but return to it in the discussion. There, using data from this study, we reconsider the criticisms of the global severity weight.

Questions To Be Answered Even in the face of these criticisms, there are a number of reasons why one may continue to use a single global severity weight in the estimation of the global burden of disease. Four of the reasons could be referred to as technical defences, i.e., they address technical deficiencies. •

There may be no variation in the average social milieu for any health condition in the disaggregated units of interest (e.g., region, sex, age, etc.), and therefore no variation in severity weight.

•

The variation in average social milieu may be so small, that it makes little difference to the outcome in the estimation of burden of disease.

•

Even if there were large variations in average social milieu, the complexity of obtaining the local severity weights would make the task Herculean.

7

•

Even if there were insurmountable problems associated with the use of an average social milieu, this aspect could be overcome by choosing an alternative definition of health based on “disability” or functional loss. A strictly functional definition could avoid the problem of variation across the disaggregated units.

The final defence will be considered in the discussion. The other three propositions require data. To date, most criticisms of the DALY have tended to be theoretical in nature and there have been few empirical studies that set out to examine the “evidence” for the DALY. The paucity of research conducted to examine the technical basis of the DALY, makes the measure extremely difficult to evaluate. The lack of evidence is, in itself, quite worrying given the eagerness with which governments are pursuing the use of the measure. In a study combining epidemiological and anthropological components, we aimed to collect data on regional variations in the severity of health conditions that would be regarded as alike, were one to apply strict criteria of “disability”. Further, the aim was to examine the degree to which variation in the severity of the health conditions could be attributed to social, cultural, gender, and environmental factors. This research was exploratory in nature. At the heart of it was the desire to examine the notion that, broadly, populations experience health conditions similarly.

Structure The remainder of the report is structured as follows. Chapter Two describes the methodology of the research, the health conditions on which the research focussed, the factors that were examined, and the qualitative and quantitative data that were collected. Chapter Three presents the results from the analysis of the data. The analysis includes qualitative and quantitative techniques. Chapter Four, draws together the theoretical arguments from this chapter, and the results of Chapter Three, and reconsiders the technical deficiencies of the DALY. The final chapter, Chapter Five, briefly outlines a research agenda.

8

NOTES 1

Murray [3] noted that the proposal of combining morbidity and mortality into a single index was proposed by Sullivan [4] 2 In the literature this is generally, though not exclusively, discussed under the rubric of the “disability weight”. We have chosen to use “severity weight” as a means of avoiding some unpleasantly tortured English that arose in our writing when “disability weight” was used. Murray and Lopez have also used an amalgam - “disability severity weight” [18] (p.378) 3 In this context the decoupling of advocacy from epidemiology related to the need correctly to count the incident cases occurring with a particular health condition. The concern was that “the squeaky wheel got the grease”, i.e., highly vocal groups could advocate successfully on behalf of their constituents for health resources even when the data were not there to support claims of need. Alan Lopez, at “Conference 2000” (Bangkok, October 2000) explained that the claims by some groups about the health resource needs often overstated the number of sufferers they represented, and therefore ‘fraudulently’ claimed additional resources. It was this form of advocacy that the DALY aimed to overcome. This is of course not a desire peculiar to the developers of the DALY. It is hard to imagine that any health care provider or indeed any advocate would endorse the idea of inflating the number of people with a health condition in order to gain extra resources. Counting the number of people correctly is a basic aim of anyone interested in population health. Another form of advocacy exists. However, it is one that the use of the DALY does not address. This form of advocacy relates to the claim that the severity of the condition has been understated, and therefore more health resources are needed. This latter kind of advocacy is far more powerful than the former and cannot be decoupled from the epidemiology in the manner that the developers of the DALY would wish. Where claims about number of people suffering from a condition can be refuted by an appropriate surveillance mechanism, the severity of a condition is always open to debate. Indeed, the creators of the DALY have taken on the role of advocates by the energy and enthusiasm with which they have promoted the DALY version of severity. The severity weights used in the calculation of the DALY represent one (widely promulgated) view among an infinity of views. 4 DALYs are potentially accrued from the moment of birth. Because a late foetal death never lives, it accrues no DALYs, and necessarily there is no burden of disease associated with the loss. Some find this counting rule offensive. 5 Although they do not directly discuss this in terms of the global severity weight, it will become clear that it may be the regional heterogeneity that is the most important criticism of the weights. 6 The use and choice of members on expert panels has been criticised because of they have tended to be made up of health care professional [29, 30]. There has, been considerable discussion about who should make the judgements [27,32,45]. 7 The health state descriptions given to the panels included a description of deafness. “Deafness – an adult whose average hearing threshold level fore pure tone stimuli of 500, 1000, and 2000 hertz with reference to ISO is less than 70 dB. Shouting at a close distance can be heard as sound but the words cannot be distinguished. The individual acquired deafness as an adult”. [2] (p.96). This of course begs the question about what the severity weight means for all those people whose clinical manifestation does not match the example. 8 In Rethinking DALYs, Murray wrote, “[w]hen making their [panel judges’] assessments, they were asked to evaluate the average individual with the [health] condition described taking into account the average social response or milieu.” [2]. Ibid.In. (p.38). There is no description of what this means. Murray noted, however, “the resulting valuations probably reflect average handicap…” (p.38/9 9 This criticism is related in some ways to the work of Davidson Gwatkin and colleagues [13, 38]. “Estimates [of the burden of disease] currently give societal averages that include data for rich as well as poor. Average figures would accurately represent the poor only if disease patterns were similar across income groups. Such similarity is unlikely, since the pattern of disease found in a population varies systematically according to its longevity” ([38], p.586-587)

9

CHAPTER 2 METHODS Aim The aim of the study was to examine the notion that the severity associated with a health condition is broadly similar without regard to country, location, gender or socioeconomic status. That is, we considered the validity of the severity weights by examining the relationship between the “experience” of a disease and the DALY estimate of burden of that disease based on the “average social milieu”. The objectives were: •

To develop detailed descriptions of the experience of the burden of disease as it relates to individuals of both sexes, in different locations, and of different socioeconomic groups; and

•

To inform policy based on measure of burden of disease/disability.

Design The study was a cross-sectional, descriptive study using both qualitative and quantitative methods to examine the impact of five factors on two health conditions. There were two levels associated with each factor: •

Country (Cameroon and Australia),

•

Gender (Men and Women),

•

Environment (Urban and Rural),

•

Socioeconomic Status (High and Low), and

•

Health Condition (Epilepsy and Paraplegia).

Each factor is discussed in turn. The impact of the health conditions were investigated using a number of different techniques, including the use of standardised quality of life scales (the SF-12 [46] and the ComQoL [47]), and qualitative techniques including in-depth interviews and Photo Voice [48-50].1 Each of these will also be discussed.

Country The choice of countries for the research emphasised the importance placed on having one developing and one developed country. In terms of our commitments and availability to work together the choice of Cameroon was arbitrary. However, the contrast with Australia highlighted cultural, economic, and developmental differences, which allowed us to explore the research question.

10

Figure 2a Map of Cameroon [51].

Figure 2b Map of Australia [51].

11

Cameroon is a small Central African nation located in the Bight of Biafra (see Figure 2a). It has a population of around 15.5 million people, 43% of whom are under the age of 15. It is typical of many developing countries, particularly in Africa, with approximately 30% unemployment and 40% of its population estimated to be living below the poverty line. It has an estimated GDP per capita (purchasing power parity) of USD$2000 [51]. English and French are the official languages, but there are a large number of different language groups spoken throughout the country. In contrast, Australia is a (geographically) large island continent with the Pacific and Indian Oceans to the east and west respectively (see Figure 2b). It has a population of around 19 million people, 21% of whom are under the age of 15. It is fairly representative of a developed country with its per capita GDP in line with other OECD economies. It has an estimated GDP per capita (purchasing power parity) of USD$22,200 - eleven times that of Cameroon [51]. English is the official and principal working language.

Gender Gender refers to the culturally and socially defined aspects of being male or female, reflecting both relations between individuals at a personal level and the values and norms that permeate the broader social structure. Gender, as a construct, is context specific, fluid, and changes over time, across cultures and socioeconomic strata. Societies create gender roles attributable to one sex or the other. There is a large body of evidence that demonstrates gender differences in the distribution of diseases within populations as well as in the experience, severity, sequelae and outcome of any given condition [52]. This comes about in part because the gender roles are differentially valued, creating gender stereotyping, inequalities and inequities. Equal numbers of males and females were included in the study to determine if the experience of epilepsy and paraplegia differs by gender.

Environment Most countries show clear differences in the health indicators for rural and urban communities. If “rurality” affects not only the prevalence and incidence of health conditions, but also affects the impact of the condition, this would have important implications for the measurement of the burden of disease (e.g., [53]). In order to examine the impact of rural and urban conditions on the experience of disease, participants were recruited purposively from both environments. The urban sample in Australia was drawn from the Melbourne metropolitan area. Melbourne, the second largest city in Australia, is the capital of Victoria, a state in the south-eastern corner of the country. The rural sample was from a range of rural communities in Victoria. Urban participants in Cameroon were drawn from the capital Yaoundé. The rural sample was drawn from the rural hinterland of Yaoundé (epilepsy) and from the central province (paraplegia).

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Socioeconomic Status Health conditions are not distributed in a society proportionately across different socioeconomic groups. The multilateral organisations refer regularly to several of the communicable and non-communicable diseases as “diseases of poverty”. In the 1999 Behavioral Risk Factors Surveillance Survey conducted by the US Centres for Disease Control for example, 10.6% of people with an income of less than $15,000 reported that a doctor had told them that they had diabetes (excluding gestational diabetes), and this percentage declined steadily over the successive income categories. Only 3.3% of people with an income of at least $50,000 reported that a doctor had told them that they had diabetes.2 In order to examine the impact of SES on the experience of disease, it was necessary to sample from people of high and low SES. Unfortunately, because many health conditions are not distributed proportionately across the levels of SES, it is frequently difficult to sample adequately from any one SES category. In cross-country comparisons such as this one, the lack of a simple means of sampling Australian and Cameroon participants from equivalent SES categories further compounded the difficulty. The difficulty was handled in three ways. First, income was used as the marker for SES. Second, participants were asked to nominate to which of five income categories they belonged. Finally, a post hoc determination was made about the SES status of a participant based on a median split of the data for their country. The (approximately) 50% of participants in the lower income categories were deemed to be of low SES and (approximately) 50% of participants in the higher income categories were deemed to be of high SES. The solution, although not ideal, made within country comparisons possible. Between country comparisons on this factor, were conducted, however, they remain problematic. For the purposes of data analysis, 41% (33) of the Cameroon participants were categorised as low SES and 54% (36) of the Australian participants were categorised as low SES.

Health Conditions In choosing a suitable health condition, one consideration was an observation made in defence of the global severity weights [43]. The defence was that cross culturally the severity weights for any health condition are stable. It concluded At the end of the day, everybody agrees that the health state associated with quadriplegia (paralysis from the neck down) is worse than the health state associated with vitiligo (patches of whitened skin) (p.16).

This is not an entirely satisfactory argument. Everybody will agree that perfect health is better than death. If there is going to be disagreement, however, it is unlikely to occur around (or near) the endpoints of the scale. It is the health conditions with a “middling” severity weight that are likely to attract the greatest disagreement, and it is here that research should focus. As an aside, a lot has been made about the level of agreement between panels about the severity of the health states. It was noted, for instance, that the agreement between different assessment panels (10 in total) about the severity of a range of health conditions was very high, even though the panel members were from divers cultural

13

backgrounds [43] (p.16). The level of agreement was demonstrated by the correlation coefficient between panels’ judgements. Because the severity scale has known and fixed end points about which everyone agrees (good health at one end and death at the other), the further one moves away from the endpoints towards the middle, the greater is the likelihood of disagreement. In other words, the level of agreement about the severity of a health condition at any one point on the scale is not constant. This introduces a problem of heteroscedasticity, and the correlation coefficient would in effect inflate the level of agreement between the panels. Returning to the study design, we were eager, therefore, to examine relatively common conditions with low to moderate severity weights. The availability of sufficient participants for the study in both Cameroon and Australia was also an important consideration in the choice of condition. The cross-country comparison effectively excluded all tropical diseases, 3 and many maternal and child health conditions. Epilepsy and paraplegia were selected as the most appropriate conditions for the study. Both conditions were symptomatic of underlying disorders and were able to capture the nature of disability, which had been identified as important in the severity weighting exercise. The selected health conditions had to be prevalent in both countries, of similar clinical severity, and readily identifiable. In Australia, for many health conditions, potential participants could be identified because of the infrastructure that exists to support people living with most illnesses. Access was somewhat more difficult in Cameroon because there was a lack of support groups and health services, and populations were highly mobile with little community infrastructure.

Epilepsy Epilepsy is a disorder of cerebral function that is characterised by a pattern of repeated, sudden brief attacks of altered consciousness, motor activity or sensory phenomena known as seizures. Epilepsy is a symptom of an underlying condition and seizures may be due to brain tumours, scar tissue from trauma to the brain, or a progressive neurological disease. In most cases however, it is idiopathic; that is, there appears to be no pathological basis for the seizures. In the United States and Canada, epilepsy is the second leading neurological disorder, behind stroke [54]. The prevalence of epilepsy is about one percent but this varies widely across populations. For example, in Africa it is estimated to lie between 0.5% and 3.7% [55] and in the United States and Canada it is estimated to be 0.5% [54]. There is a significant level of under-reporting because of the stigma associated with seizures [56]. There is also some indication that some sub-groups have a genetic predisposition to lower seizure thresholds and consequently a much higher prevalence than is reported within their general populations. According to the Global Burden of Disease study, an untreated seizure disorder has a severity weight of 0.15 in adults, and a treated seizure disorder has a severity weight of 0.065 [45] (p.413).4 In order to compare, as much as possible, conditions of like severity, inclusion criteria were applied to potential participants in Australia and Cameroon (Table 2.1). Despite the intention to do so, these criteria were not uniformly applied. Consequently, in Cameroon for instance, while participants may have been prescribed medication, they were not necessarily taking it regularly because of lack of availability or accessibility. Similarly, there were two participants in Australia who were not on medication at the time of the study but had recently had lobectomies performed which largely controlled their seizures.

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Table 2.1: Inclusion criteria for participants in the epilepsy component of the study. • • • • † ‡

•

Age 25-35 Epilepsy (“Grand Mal”) for at least five years† Living in the community Frequency: less than 1 seizure per day and more than 1 every 2 months On medication‡

The criteria of “Grand Mal” seizures meant that witnesses could externally verify the occurrence of a seizure. This was particularly important for the inclusion of participants from the Cameroon, some of whom had not seen a doctor. The medication criterion was to ensure that both Cameroon and Australian participants were being treated for their epilepsy.

The Australian participants with epilepsy all came from the south-eastern state of Victoria and were largely self-selected through the Epilepsy Foundation of Victoria. With the support of the Executive Board of the Foundation, a notice was placed in their quarterly newsletter, Headlines, with an additional note from the editor endorsing the aims of the study. Potential participants contacted members of the research team for further details and were then recruited into the study. Fifteen (15) males (mean age =29.6, sd=5.9) and 19 females (mean age 30.5, sd=6.1) participated in the study.5 Table 2.2 below shows the distribution of the male and female participants with epilepsy between the city of Melbourne (the urban area) and the rural areas of Victoria.

Table 2.2: Characteristics of the Australian participants with epilepsy Urban

Rural

Total

Male

8

7

15

Female

13

6

19

Total

21

13

34

The Cameroon participants with epilepsy were identified from one of two sources. The urban sample was identified through a Catholic Health Centre in Yaoundé (Centre de Santé Mental Benoit). Potential participants were contacted directly by the research team and asked if they would be willing to participate. Most of the rural sample was drawn from villages along the Sanaga River north of Yaoundé – an area known among local health workers to have a high prevalence of epilepsy. Villages were then visited directly by members of the research team. Two rural participants were identified by traditional healers in Yaoundé and directly approached by members of the research team. In all twenty-three (23) males (mean age =24.5, sd=4.9) and 19 females (mean age 24.2, sd=3.4) participated in the study. Table 2.3 below shows the distribution of the male and female participants with epilepsy between the city of Yaoundé (the urban area) and villages along the Sanaga River (rural area).

15

Table 2.3: Characteristics of the Cameroon participants with epilepsy Urban

Rural

Total

Male

12

11

23

Female

8

11

19

Total

20

22

42

Paraplegia Paraplegia describes paralysis of the lower portion of the body and of both legs, usually caused by a lesion involving the spinal cord as a result of trauma, a congenital malformation or a disease process. The average disability weight for paraplegia, from an estimation exercise conducted in Geneva, was 0.671 [2] (p.33).6 Again, inclusion criteria were applied to potential participants in Australia and Cameroon (Table 2.4), and again, despite intention, they were not uniformly applied. There were three participants from Cameroon and one from Australia who were diagnosed with paraplegia and had limited mobility on crutches.

Table 2.4: Inclusion criteria for participants in the paraplegia component of the study. • • • • •

Age 25-35 Loss of the use of both legs from spinal cord damage Paraplegia for more than 5 years Living in the community NOT Polio*

* Preliminary attempts to ascertain people with paraplegia in Cameroon resulted in health care workers wrongly identifying people who had polio as paraplegic.

Australian participants with paraplegia all came from the southeastern state of Victoria and were largely self-selected through ParaQuad (Paraplegic and Quadriplegic Association Victoria) and the Wheel Chair Sports Association of Victoria. Permission was obtained from ParaQuad to place a recruitment notice in their newsletter, with endorsements from the editor to encourage participation in the study. Other potential participants received the information about the study through snowballing from a community medical practice. Participants in the study were, again, self-selected. Potential participants contacted the research team for further details and were then

16

recruited into the study. Nineteen (19) males (mean age=34.5, sd=4.4) and 17 females (mean age=32.8, sd=4.0) participated in the study. Table 2.5 below shows the distribution of the male and female participants with paraplegia between the city of Melbourne (the urban area) and the rural areas of Victoria.

Table 2.5: Characteristics of the Australian participants with paraplegia Urban

Rural

Total

Male

10

9

19

Female

11

6

17

Total

21

15

36

The Cameroon participants with paraplegia living in Yaoundé (the urban area) were identified through The Centre for the Rehabilitation of the Disabled at Etougebe and disabled associations such as FACAPA and HANDISPORT Cameroon. The rural sample was drawn from the central province. Local rural hospitals and disabled associations identified potential participants. In all twenty-one (21) males (mean age=33.9, sd=7.9) and 19 females (mean age 32.1, sd=6.4) participated in the study. Table 2.6 below shows the distribution of the male and female participants with paraplegia between the city of Yaoundé (the urban area) and villages in the central province (rural area).

Table 2.6: Characteristics of the Australian participants with paraplegia Urban

Rural

Total

Male

10

11

21

Female

10

9

19

Total

20

20

40

Data collection One of the Australian team (D.R.) visited the research team in Cameroon in February 2000, during which time the final decision was made on the specific disease conditions and the research protocol was finalised to ensure that similar techniques

17

would be used in both countries. Data collection occurred between March and October 2000. Data collection followed a substantially similar pattern in Cameroon and Australia and was completed in two phases. In Phase 1, the research assistants engaged each participant in an in-depth interview following the structured and semi-structured parts of the research protocol.7 In Phase 2, participants who had agreed to participate in the Photo Voice exercise were reinterviewed using their developed photographs as interview guides. The phases are discussed in turn.

Phase 1: Interview The principal interviews, in which all participants engaged, followed a standard pattern: •

Demographic questions;

•

Disease history questions;

•

ComQoL Scale;

•

SF-12 Scale;

•

Control, Optimism, and Self-Esteem questions; and

•

Semi-structured interview.

All interviews were audio taped. Interviews in Cameroon were conducted in English, French, or Pidgin, depending on the language in which the participant felt most comfortable, and these were subsequently translated and transcribed into English. Interviews in Australia were conducted in English with participants who were fluent English. Half the Australian protocols were fully transcribed and half were supported by detailed field notes. The Australian protocol was used as the reference protocol and the final protocols were agreed upon at the meeting in Yaoundé in February 2000. The Cameroon research team, using a translation and independent back translation model, developed the Cameroon English, French and Pidgin translations of the protocol. The Cameroon English and the Australian English protocols varied to take account of differences in the idiomatic expression of spoken English in the two countries. For example the phrase “could not be more important” was rendered into Cameroonian English simply as “very, very important”. Similarly, the demographic questions varied slightly to take account of structural differences between Cameroon and Australia. For example, in the Australian protocol the question about marital status did not include the option for polygamous marriages that was included in the Cameroon protocol. The question about income differed in the unit of currency and in the scaling of income between the two countries. Questions about the history of the disease had the dual purpose of (a) ensuring that participants from any single health condition had, as much as possible, the same level of functional loss, etc., and (b) it collected, background information. The ComQoL or Comprehensive Quality of Life Scale is a highly researched and cross-culturally applied instrument for measuring subjective quality of life [47]. Subjective quality of life is measured with 14 questions. Two questions are asked in relation to each of seven domains, one concerning the level of domain satisfaction and the other concerning domain importance. The seven domains have been proposed on

18

both theoretical and empirical grounds [47], and cover material possessions, health, and achievement in life, relationships, safety, community, and happiness. The summation of the “satisfaction” items was used as the measure of subjective quality of life and can be referenced to an international “gold standard” [58]. The SF-12 is a standardized and widely used scale in health research with welldocumented methods regarding its use, validity and scoring [46]. It is based on the longer SF-36, and measures health-related quality of life (HRQoL) in two dimensions, mental health and physical health [59]. It has been widely used in cross-national studies and found to be a useful measure of HRQoL. The version used was based on the Australian translation of the SF-36 [60]. The scoring of the SF-12 was conducted according to the US scoring manual [46]. However, because the interest was in comparing the participants across the separate factors, it was less critical that there was no locally applicable reference standard. Ten broad general questions were asked in the semi-structured interviews such as “What impact has it [paraplegia or epilepsy] had on your life?” The impact that the explanatory factors (gender, environment, and SES) may have had on the experience of the disease was captured in part by questions such as “What difficulties do you think that you experience from epilepsy/paraplegia because you are a man/woman that the opposite sex would not experience?” All interviews were audiotaped. The Cameroonian interviews were fully transcribed. Approximately half the Australian interviews were fully transcribed and half had detailed field notes made of the interviews to support the audio recordings. Photo Voice is a technique that enables a compilation of visual images generated by the participant that provide insight into the factors that create their reality [48-50]. Thirty participants in Cameroon sampled from all levels of the factors (health condition, gender, environment, and SES) and 20 participants from Australia were provided with disposable cameras and instructions on their use. Participants were asked to take photographs around their daily tasks, particularly focusing on how their health condition affects their activities. These included images that represented their activities of daily living as well as images that represent things they were unable to do as a result of the health condition. Participants were asked to select six of the developed photographs (three around daily living tasks, three around limitations) and these were used as prompts in a second in-depth interview.

Data Analysis This study was principally exploratory in nature and heavily reliant on qualitative methods. If social, environmental, and cultural factors would affect the experienced severity of a health condition, the qualitative data and their analyses should speak directly to the question. The analysis of the data was driven by the need to identify themes that were common to people with a particular health condition in one context versus those in another context. Exploratory and descriptive data analytic techniques were used in the analysis of the SF-12 and ComQoL data. The principal focus was on linear associations between the scale measures and the study factors. The analysis of the scale data was also used to inform the analysis of the interview data, and vice-versa. Thematic analysis of the qualitative data including the data from Photo Voice was the principle approach to the qualitative data.

19

The analysis of the ComQoL data focused on the satisfaction scale. This was made up of the questions related to how satisfied a participant was with the seven domains. The mean score of the satisfaction scale was used as the general measure of subjective well being. The analysis of the SF-12 data focused in part on the mental health (MCS) and physical health (PCS) sub-scales [46]. However, in some cases the level of missing data was too high. Three scale items within the SF-12 used five point Likert-type scales dealing with self-rated health, pain, and physical and emotional health. These items were well completed by participants, and were analysed separately. The quantitative data were analysed in STATA and SPSS. Descriptive statistics, Student’s t-tests, and analysis of variance (ANOVA) were the principle techniques. For each health condition the factors of the study (country, gender, SES, and environment) were used as independent factors. The dependent measures included the mental and physical scores on the SF-12, some individual item scores on the SF-12, and ComQoL scores.

Ethics The research was passed by the Human Research Ethics Committee at The University of Melbourne, Australia and by the Research Ethics Committee at IFORD in Cameroon. All participants gave consent to their participation following a full description of the purpose of the study and the nature of their involvement. Separate consent was obtained for Photo Voice and for the use of the photographs in the study. There were no adverse outcomes anticipated from the study and none were recorded, with the exception of one Australian participant who withdrew from the study following the occurrence of a dream of ill omen.

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NOTES 1

In the study protocol, items were also included about control over life, self-esteem, and optimism. Analyses of these items, however, are not presented in this report. 2 There are clearly problems with relying on self-reported conditions as evidence of an actual health condition. Nonetheless, for demonstration purposes, this provides an easily verifiable example of difference in prevalence by SES: http://apps.nccd.cdc.gov/brfss/income.asp?cat=DB&yr=1999&qkey=1364&state=US 3 Although Australia does have a tropical zone, most serious tropical diseases (such as Malaria and Schistosomiasis) are not endemic. Furthermore, most of the population does not live within the tropics, further reducing their exposure to tropical diseases. 4 The Australian burden of disease study used a slightly different severity weight (0.11) 10. Mathers C, Vos T, Stevenson C. The burden of disease and injury in Australia. Canberra: AIHW; 1999. (p. 187) based on weights from a Dutch study [57]. 5 There were originally 20 female participants with epilepsy in the Australian study. One participant withdrew from the study citing a premonition from a bad dream. All data relating to her were expunged. 6 In the Australian burden of disease study, “spinal cord injury” attracts a severity weight of 0.725 [10] (p.201); however no distinction is reported about the level along the spine at which the injury occurs. Murray [2] records an average severity weight of .895 for quadriplegia and .671 for paraplegia (p 39). 7 The protocols are available on request. The Australian protocols are available in English, and the Cameroon protocols are available in English, French and Pidgin.

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CHAPTER 3 RESULTS The results are divided into two broad sections. The first section presents largely descriptive qualitative data from the participants in Cameroon and Australia. The section is supported, however, with some descriptive analyses of the quantitative data. Where appropriate, sections of interview transcripts have been provided in “text boxes” as a means of contextualising the data. The images in the plates come from the participants’ Photo Voice activity. The second section is largely quantitative, exploring how the factors of country, environment, gender, and socioeconomic status (SES) relate to the health-related quality of life (HRQoL) measures (i.e., the SF-12) and the subjective quality of life measures (i.e., the ComQoL). Although the factors are discussed separately, it should be noted, particularly when reading the qualitative analyses, that the factors are closely interrelated. It is not possible to consider gender for instance as discrete from SES, or the environment in which one lives as independent from SES. It should also be remembered that the examples are illustrative rather than definitive, and such observations simply reinforce the sense that the interactions are complex.

Paraplegia Participants in this sample of the study became paraplegic mainly through injuries sustained as a result of road traffic crashes, work and recreation-related falls and violence including two gunshot wounds and physical assaults. Four participants reported medical conditions: two from Cameroon with a spinal tumour and spinal tuberculosis and two from Australia with acute myeloid leukaemia. In general, most of the participants described trying to cope with their disability and at the same time, having to fight against a society that perceived them as lesser human beings. Examples given for this perception included lower paying occupations for the disabled as well as being paid less than the able bodied in various other jobs. In broad terms, however, the descriptions of the effects of paraplegia were very different for the two countries. One of the obvious distinctions between participants in Australia and Cameroon was the possession of a wheelchair. Everyone in the Australian sample (n=36) had a wheelchair as a result of government policy to ensure the provision of this aid to anyone with severely impaired mobility. Of the 40 people with paraplegia in the Cameroon sample, 32.5% (n=13) did not own a wheelchair. Fifty percent of those living in a rural area (10/20) and 15% of those living in an urban area (3/20) were without wheelchairs.

Paraplegia in Cameroon Data from the Departments of Health Statistics, Community Health and the National Centre for the Rehabilitation of the Handicapped in Cameroon indicated that at the primary level, people with paraplegia were treated at physiotherapy centres in

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provincial hospitals in the country. If they survived and were in need of follow-up and rehabilitation, they were sent to specialised centres like the National Centre for the Rehabilitation of the Handicapped (CNRH) in Yaoundé, Saint Joseph's Child and Adult Home (SAJOCAH) Bafut, and Mbingo Baptist Hospital in Kom North West Province of Cameroon. 1 The Mbingo centre was a community-based centre whose patients have home-based care. SAJOCAH and CNRH were initially set up as centres to promote physical and occupational therapy of the handicapped. The occupational therapy program of the CNRH has ceased due to lack of funds. The Mbingo Baptist Hospital is the only facility that currently provides occupational therapy. Places are limited in all these facilities and priority is often given to those who can afford to pay more than the prescribed rates for the services. None of the participants in the study were under the care of any of the above institutions and none had access to professional care at home. All participants in the study reported problems with managing varying degrees of urinary and fecal incontinence and all reported decubitus ulcers (pressure sores). The other major health concern reported was pain. All paraplegics get these sores because their bodies are inactive. Then they start rotting in the back and in the buttocks. They can’t even be cured in the general hospital.

Gender factors in paraplegia in Cameroon For women with paraplegia, domestic activities were severely restricted – they were unable to deal with domestic chores such as doing the laundry and cooking unless there was someone to put everything within their reach. Women reported that in general, it was more difficult for them to organise carers. Six women were single parents and were still responsible for maintaining a household – they had been deserted because of their handicap. With restricted earning capacity, financial resources for basic living and health care for themselves and their families were a major problem. In Cameroon women are traditionally responsible for feeding the family, which includes the purchase of food items from the markets. This activity was one that all female participants reported as being impossible (even for those who did have access to wheelchairs) because of the congestion at the markets and lack of access. The restriction in domestic activities was also problematic because a lot of the cooking was done over open wood fires. Problems arose from the inability to obtain and chop firewood, and the potential for serious burns. Plate 3.1 portrays the cooking facilities of one of the betteroff women living in Yaoundé. Although she did not have to worry about firewood, there are clear safety issues in the set-up of the kitchen. She had to reach up to the pot, could not look into it, and had very little control over the stovetop.

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Plate 3.1 Cooking with paraplegia in Yaoundé

Faecal and urinary incontinence was an issue. It was exacerbated for both men and women by the difficulty in maintaining personal hygiene and it was particularly problematic for women during menstruation. This was largely due to a lack of access to pipe-borne water; washing was from a bucket and required getting the bucket of water to the bathroom (see plate 3.2). Personal hygiene problems had a significant effect on selfesteem and consequently, the formation and maintenance of intimate relationships. Plate 3.2 Negotiating obstacles at bath time

Women in the Cameroon sample also raised the issue of reproductive health, although the responses to the issue varied. Some women objected to the expectation that they would continue to carry out reproductive roles and have intercourse with their partners.

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Women still have to have sex if they are still married and their husbands are taking care of them because men are like that whether a woman is handicapped or not, men don’t care, they still have to have sexual contact with them. Carrying a pregnancy and to be creeping on the ground is very bad and difficult. Concerns expressed also included the potentially embarrassing situation of urinary and faecal incontinence during intercourse and the perception that they were repulsive. This idea that they were repulsive gave rise to a further sense that they were being used without actually being engaged as individuals in the act of intercourse. When I have sex, I worry very much because when he mounts me, I might start feeling like I have to urinate or pass out∗ . But you have to make every effort to encourage your man whether in this sense or any other way.

I became paraplegic because my husband beat me. He never regretted battering me and continues to insult and use me because I am now handicapped. I am still staying with him and he treats me very badly, but look at me, if I leave him now, who will take me? A few thought that their ability to continue to have intercourse in spite of their injuries gave them an advantage over males with paraplegia who were impotent as a result of the disability. The women with children (n=10) expressed concern about the safety of their children. The mothers had to rely on others to manage much of the day-to-day childrearing responsibilities, and this responsibility frequently fell on the eldest children who were often only slightly older than their younger siblings. Men in the sample described in some detail why ‘sexual incapacity’ was the ‘first and most important burden’ they faced. They found it difficult to cope with the challenge of a ‘diminished manhood’ and their inability to find or retain a wife who would look after them. As concerns sex, I am no longer a human being. Now I am just like a dry stick; a skeleton. I can’t have intercourse again. Men also spoke about the loss of their role as the breadwinner and the patriarchal figure within the home. Because of this situation I am in, I cannot get money and you need money to marry a woman. And even if I did have money, I cannot service her because I am not a man any more. I was abandoned by my wife because to her I was not a man, no money, no nothing.

∗

defaecate

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Economic factors in paraplegia in Cameroon A person’s economic situation is a determinant of many health conditions. The poorer one is, the higher the risk for a variety of health conditions. Similarly, a person’s health condition is a determinant of their economic situation. The sicker one is, the more expensive the treatment and the less able one is to work. The reflexive nature of the effects of participants’ economic status on the experience of paraplegia as well the effect of the paraplegia on their economic status was evident in the qualitative data. Participants who were relatively well off in Cameroon were able to purchase wheelchairs and pay for some support and assistance. There was also a wider range of options for them in terms of career choices, with one able to access computers and run a home business and another working as a disc jockey. In general, however, the lack of general infrastructure to support the condition, such as wheelchairs and appropriate means of public and private transportation, meant that their mobility remained severely restricted. Only one participant had her own means of transportation. The rest of the participants had to rely on others or were confined to their homes. Transportation with taxis was four times more expensive because the drivers demanded compensation because (a) the disabled passengers reduced the drivers’ opportunities to obtain additional fares through sharing the taxi with other passengers, and (b) the drivers had to assist the disabled in and out of the taxi. The construction of disability-friendly homes was not possible because of the lack of expertise and financial constraints. Wheelchairs available in Cameroon were manual and very rudimentary. Most of the participants needed to be pushed around and could not move themselves because the manoeuvrability of the wheelchairs was limited. This task often fell on young children who were relatives or domestic help (see Plate 3.3 and 3.4). Left Plate 3.3 Children called on to push wheelchairs Right Plate 3.4 The ‘Tricycle’

Most participants were unable to afford basic health care to improve their quality of life, such as treatment for incontinence, and reported being treated very badly by the health services. For those in rural areas, the hospital was typically 17-20km away, making it an expensive journey, even before the actual health care costs were taken into account. Mothers in general and single mothers in particular with young children described their desperation and frustration in trying to cope with illnesses in their children. The participants also considered themselves to be a major burden on their families, as increased resources had to be spent on their care and treatment. Most of the rural sample had engaged in subsistence farming or fishing prior to their injuries and could no longer engage in these activities. They reported that there seemed to be an

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expectation that handicapped people would become involved with craft activities like basket weaving; … but even if you did that and were selling the products, after a hard day’s work, you still got treated badly like a beggar and not like someone with any importance to the community.

I used to engage in petty trading. I have not got the courage to go out and battle with people. This is because of the problems that I have. You should know that when I want to urinate, I cannot retain it for more than five minutes, maybe three. I have to put on something to retain the urine but that means I smell. So better I stay home.

If I had money, I would get a car so that I can get out a bit more. Not seeing the sun is very depressing. I know money will not get me new legs but perhaps with my own vehicle I can change my surrounding a bit.

I would send my children to school if I had money. My family have now become victims of my circumstances and my daughter is thinking of getting out of school even though she is very clever but I can’t buy her books or her uniform.

Money? Money is only of value to people who have legs Two women from the rural participants were able to continue their occupations. One was a hairdresser specialising in hair braiding. The other was a street vendor whose main problem was her inability to chase away ‘shop lifters’, but she was otherwise pleased to be occupied and earning some income.

Environmental factors in paraplegia in Cameroon Environmental conditions for people with paraplegia in Cameroon were almost uniformly inadequate both in urban and rural centres. The SF-12 showed a significant difference in the physical health scores between people with paraplegia who lived in urban versus rural environments (t=3.307, df=32, p=0.002), with those in rural environments having lower scores. This difference was also clearly demonstrated in the qualitative data. The various qualitative factors that contributed to environmental problems included poor road infrastructure, poor facilities for the disabled both within the home and the community and inadequate health services. Roads in most parts of Cameroon, even in the capital city were often in disrepair and pavements for pedestrians were generally unsuitable for wheelchair access. The use of the wheelchairs on the poorly maintained surfaces also reduced the life of the chairs.

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The situation was worse in rural areas where roads were untarred. Access to homes was via paths marked only through constant use, and these were often impassable even for able-bodied pedestrians during the rainy season (see Plate 3.5). The hills were an impediment, even for those who had wheelchairs; none of the participants could afford or had access to motorised chairs. Plate 3.5 Road access in a village in rural Cameroon

Our compound is surrounded by hills and we are almost at the top of this hill. When I have to walk down there, I have to sit on the ground and crawl on my tummy and then roll on my buttocks Those who were employed or engaged in some economic activity often had to stay home during the rainy season because of the poor condition of the roads, which had implications for their income. When the road is bad like this even the person pushing me complains a lot and I have to beg him all the time. Going out is not possible until the dry season. And when it rains like that my customers cannot come for me to repair their watches and radios and so how can I eat. Even in the dry season sometimes the road becomes like an industry where dust is produced. Homes, shops, offices and other personal and community spaces in urban areas were not adapted to people with disabilities. This was attributed to a lack of expertise, resources, and community will. Participants in the city complained about their homes having elevated verandas and staircases. Rooms were small and could accommodate little else besides the wheelchair; doorways were often too narrow for the wheelchairs to get through. In many African countries, large family groups share accommodation with several individuals in one room. Cameroon was no exception and under these conditions wheelchairs were extra furniture that often could not be accommodated.

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Sanitation facilities for people with paraplegia in rural areas were generally very poor. Toilets in rural areas were largely public and required squatting, which was a problem, particularly with pressure sores. Access was also very difficult. The smell of the toilets meant that they were often located more than 100 metres away from homes. This made access difficult and during the rainy season wheelchair access essentially impossible. Three quarters of the participants described having to crawl over the ground or requiring assistance to get to rudimentary toilet facilities. Some toilets were constructed with wood planks placed over a hole in the ground and it was not uncommon to hear reports of people falling through the decomposing timber. (Plates 3.6, 3.7, 3.8)

Plate 3.6 Most accessible rural toilet for people with paraplegia

Plate 3.7 Example of a toilet in villages in Cameroon

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Plate 3.8 Access to a community toilet in rural Cameroon

It takes me about 5 minutes to get to the toilet. I take the wheelchair until it get there and then I crawl until I am close enough. And then I lie down on my back and adjust my buttocks until my anus is facing directly in the hole and then I can use the toilet. Within the urban areas, only one participant reported having adequate wheelchair accessible toilet facilities. (Plate 3.9) About a quarter of the participants had made arrangements in the house to use local commodes (see Plate 3.10) or ‘cholera beds’ with pots, which they had to clean themselves or relied on carers to clean. Participants often remain soiled, particularly if incontinent, and this again predisposed them to pressure sores and infection. The odour led to further stigmatization and isolation.

Plate 3.9 A toilet adapted in an urban home in Yaoundé

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In this room I do every thing, it’s a kitchen, bedroom, sitting room and toilet. Because I have no control, they have made a hole in the middle of the bed so whether I am lying down or sitting up in the bed, it passes through and into a pot under the bed. Because the room now smells very much, I don’t have anybody come and see me.

Plate 3.10 Home made commode in rural Cameroon.

In general, most of the participants felt that the disabled were better off in urban areas because there were more people who were prepared to offer assistance. A small number (4) of the urban participants thought that they would have been better off in a rural community because of the stigmatisation in the city and the general impression that all disabled people were beggars. One, however, described a friend whom he had met during rehabilitation. The friend had left town to a rural area for better family support but had died within a month.

Other social sequelae of paraplegia in Cameroon Two thirds of the participants lost their able-bodied friends following their injuries due to a general belief that they were in some way responsible for their disability. Locally, it is believed that those who become disabled have wronged someone personally, are being made to atone for the wrongdoing of a close relative, or have indulged in witchcraft. Their presence is therefore considered a bad omen. Intimate relationships were generally affected; they were discriminated against in the workplace, paid lower wages and treated as “charity cases”. They were insulted in the streets because they were assumed to be beggars and were cheated by vendors.

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The isolation that resulted either from mobility problems, stigmatisation or low self-esteem was discussed as a potential reason for suicide. Six participants reported that they regularly felt like ending their lives because they did not “get to see the sun” (an expression describing both social isolation and confinement)

Paraplegia in Australia There are a number of organisations in Australia that support people with paraplegia. The ParaQuad Association provides a wide range of services from personal care and facilitation of community access to support services like respite care. The Wheel Chair Sports Association also provides both a supportive role and rehabilitation through sporting activities. The disabled who are not in full time employment are means-tested and are supported through social welfare disability pensions and subsidised goods and services. In addition, depending on the cause or location of the accident, there is a range of insurance schemes to which individuals or their employers belong that provides financial support following injury. Government provides some assistance for equipment such as wheelchairs, and most buildings and public spaces are required by law to ensure adequate access and safety for the disabled. A general characteristic of the Australian sample was their interest in sport, fuelled in part by the strong promotion of wheelchair sports in 2000 in the lead-up to the Paralympics 2000 held in Sydney. In Australia, sport is the great leveller. The disabled gain their respect in the community through this medium and the community also get to learn more about the disabled and hopefully gain a bit more respect in the process. Due to the nature of sampling, some participants were highly motivated achievers who reported that they had accomplished more with paraplegia than they would have done if they were able bodied. There were, for instance, participants who had taken up road racing, paragliding and bungee jumping as hobbies since their accidents. However, although there were a few who reported coping very well with the disability, most raised issues that shed light on difficult health-related experiences. Uncontrolled pain and spasms were a common complaint. Others included urinary tract and renal infections particularly amongst participants with in-dwelling catheters and some liver problems. In general, it was reported that health services consistently ignored or failed to appreciate the magnitude of these problems. There was also a perception of stigmatisation in general but particularly within social welfare, health services and the medical profession. We get second-class doctors, second-class nurses and second-class care. They throw us in the corner and hope we will go away.

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The doctors I had did not allow me to be hopeful. They kept insisting that I seek counselling to learn how to cope with the fact that I would never walk again. I was already trying to cope with the fact that I was driving the car for this accident and that my sister lost her life. And I am confined to a wheelchair, sitting here 18 hours a day and depend on other people and they [the doctors] don’t even want me to try.

I find that the doctors do not see you as a whole person and they are only interested in treating individual symptoms. They will talk about nerve damage or broken bones, but don’t tell me anything about how to cope with the constant pain or what to do about my bowel and bladder issues or pressure sores.

The hospitals now want to push you out so fast that they don’t even warn you about what to do about pressure sores. I had someone else teach me how to weight change. We just don’t get to learn how to cope properly. Half of the participants were smokers, with all but two taking up smoking following their injury or disability. The reasons for smoking varied from “needing to do something with their hands” to “a relatively harmless way of coping with stress”. The national average of smokers in Australia is about 20%.

Gender factors in paraplegia in Australia The qualitative gender factors for the Australian participants highlighted some problems in fulfilling gender roles and the need for dependence on others. Both men and women talked about sexuality but with a different focus. The issues for women in intimate relationships related to self-esteem and their perception of being sexual beings. Sometimes with a catheter, I have accidents when little bits leak. I remember on many occasions I’d have a leak on the ground and he’d step in it in his socks and he would blast me, really blast me. And I would apologise and apologise and try and explain that it is not my fault and he would say I don’t care, it still pisses me off. And he would get angry and it would get to the stage I’d really think he was about to lash out. I felt so bad about myself I started punishing myself. And eventually I thought I’ve had enough of this. And the house was made up for me and the chair so I made him pack up and leave. Now if I have a leak I know – hey, it happens.*

*

This excerpt from the transcriptions describes a situation very similar to a case reported in Cameroon data (see page 23). The major difference is that women in Australia have practical alternatives if they choose to leave abusive relationships because of the existing structures to support abused women in general and other support for disabled women.

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When I first met him, I had the catheter; I often had bowel accidents, which are very unpleasant when you are trying to be sexy. But he was very supportive. It was very scary, I can’t feel properly anymore, but it was okay and when we did have an accident, I would think, oh yuck, but he would pick me up and wash me. He made me stop putting myself down. All the women with children had learned to cope with childcare. One participant explained how her children had learnt at a very young age to sit still while they were on her lap so that they would not fall off and another described playing with her nieces when she was minding them. The biggest problem is dealing with other people in the community rather than my inability to cope. The other day my twin nieces bolted when I was with them in the shops and we were having a jolly old time playing hide and seek when this woman who thought she was helping grabbed them up in her arms and delivered them to me with a stern ticking off about not running away from me. We were doing fine, I didn’t want her to help and she could have asked me first. Others found that they were treated as incompetent mothers because they were in a wheelchair. I come across a paediatrician who must have thought I did not have a head maybe I had two heads. My first son was milk intolerant and for six weeks he was just crying and I honestly thought it was a normal thing – babies cry and mum kept saying he will settle down but I was worried so I took him to see this doctor who said he had to be admitted in the hospital without saying why. I got really upset because no one would tell me why and I was crying and told mum they think I am an unfit mother. She said these things happen, you just have to find the right person to talk to. So I went again to the hospital with my husband and they did not speak to me, just directly to my husband. And all they to did was explain to me that my son had to be put on soymilk. I was such a mess and they still would not let me take him home … I decided after a few days that I would read his chart and they stated in there that the mother has bonding problems with her child … I finally talked to the doctor after chasing him for 2 weeks and said that obviously because I am in a wheelchair I cannot hold the baby enough… and then I heard him say to one of the nurses ‘poor child, not only a young mother who does not know what she is doing but also in a wheelchair. Well I took him home after that and we have done very well. He is four years old now and he loves a cuddle and a ride on the chair. There was a very strong expressed need for independence by both men and women, but the effects of lack of control were expressed more strongly by male participants.

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I had my own life, doing my own thing and all of a sudden I am no better than a baby who needs everything done for him. I really loathe it A common issue raised by the male participants was sexuality and developing and maintaining intimate relationships. Some reported that women were very sympathetic to their predicament with one participant describing his wheelchair as his “chick magnet” and another as his “icebreaker”. However, most reported that the wheelchair was a “turn off” because of the assumption that physical disability also implied intellectual disability.

Economic factors in paraplegia in Australia Qualitative data on the economic factors were equivocal. Three participants reported that they became better off economically as a result of their injuries because they had received lump sum payouts from insurance companies. This meant that they were able to improve their standard of living compared to the pre-injury state as well as to being able to buy the goods and services needed to facilitate their functioning with the disability. It was also clear that with financial resources, the technology was available to enhance functioning - ranging from transportation to labour-saving devices within the workplace and the household. However, payments were often delayed and usually provided as a pension rather than a lump sum payout. The implication of this was that participants affected did not have the necessary capital to adapt their surroundings to the disability. There were also a few reports of exploitation and abuse by friends, relatives and / or other support people who were supposed to provide assistance. The main economic issue with paraplegia described by most of the participants was the curtailment of earning capacity. None of the participants was able to return to jobs they had prior to the injury. Participants who had sustained work place injuries in the construction and mining industry, for instance, could no longer return to work and although they had received some financial compensation, they were currently unemployed, socially isolated and expressed concern about depression. Four participants had taken up competitive wheelchair sports following their injuries and earned national and international recognition for their athletic abilities. All reported that they did not believe they could have achieved as highly if they had not found themselves in their current situation. Another participant who had sustained the injury through violence reported that without the injury and the need to re-evaluate the direction of his life, the likelihood was that he would have ended up in jail or dead. Three participants were in middle to top management positions. Two of these were from organisations set up specifically to advocate on behalf of the disabled.

Environmental factors in paraplegia in Australia The qualitative data reveal some differences between participants in rural and urban areas. Participants in rural areas reported less infrastructure that supported wheelchair access and activities that were inclusive of people with disabilities. There was also less support and services for people with paraplegia in regional centres and hospitals. Participants in rural communities found they had to restrict where they went depending on whether or not the venue was wheel chair friendly.

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Our local shopping area has maybe 10 shops – I can’t get into eight of them so I stay outside until someone comes to ask what I need – sometimes they do and sometimes they don’t. So I wait until I have the opportunity to go into town; it is very restrictive.

Other social sequelae of paraplegia in Australia Stigmatisation was commonly reported as being of concern and as mentioned above, the effects of this were manifested in employment opportunities, access to quality health services and access to community facilities. When I ask a question in a shop and I am with my wife, they provide her with the answers. We went to buy a car and I asked all the questions but they only spoke directly to her. She knows nothing about cars but because I am in a chair, they assumed I was too stupid to understand what was going on.

The woman across the road, who used to be a friend of mine said “If I was in your position, I’d kill myself. What do you have to live for?”

Some see a wheel chair and think no legs, no brain. I have had people shout at me because they thought I was deaf as well.

Basically, we live in a world that is set up for the able-bodied and not the disabled and we have to learn to live in it and view things from that perspective. In that sense, those who come late to disability have a better chance of understanding and coping I think than those born with a disability.

People in the community think the disabled have it easy because of payouts. I get comments like ‘he gets special treatment because he is disabled’ or ‘the only reason he got where he is, is because he is disabled’ Participants with families found they could not always assume they would have the help and support they would need to cope with the condition. Some described how much harder their parents or partners took their disability than they did.

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They desperately needed someone to blame, there was a lot of pity and so much anger. By the time my mother died [of cancer], she was blaming me; it was my fault that I had an accident and why she had to stay home. She started drinking a lot, our relationship went down. She said if it were not for me she would be out there working. I don’t really blame my dad, he had to cope with mum as well – she became a very depressed person. But my sister – she was expecting a baby when it happened and she came to see me in hospital. She would cut off her right arm for me; just one of those once in a lifetime people.

Epilepsy Most cases of epilepsy were idiopathic in that they were not associated with any other known injury or pathology. Five participants in Australia reported that their seizures followed meningitis, a tumour, a cerebrovascular accident and a complicated pregnancy. None of the participants from Cameroon was able to report any specific pathology that could have been related to their seizures and most attributed their condition to supernatural forces including bad luck, jealousy, witchcraft, and divine punishment. One of the objectives of this study required the comparison of people with similar clinical presentations of epilepsy. For this reason, there were inclusion criteria about the minimum length of time the participant had epilepsy, their age, the frequency of their seizures. Checks were included in the protocol, such as asking about the duration of time since the last seizure, but it was not possible to screen the participants clinically. Figure 3.1 shows, for Australia and Cameroon, the duration of time, in days, since the last seizure. It is apparent from the graph that the seizure frequency was much lower among Australian participants, suggesting better-controlled epilepsy. Surprisingly, the median time since the last seizure was the same for both the Australian and Cameroon sample, namely 30 days. There were no significant differences between those participants who had a seizure within the last thirty days and those who had not, on the SF-12 subscales or the ComQoL.

Epilepsy in Cameroon The Cameroon public health department has not yet carried out any action against non-communicable diseases (NCDs) in general and epilepsy in particular. Policy document reviews and key informant interviews revealed that NCDs are on the rise and authorities recognise their emergence as a major health problem. However, they have not been included as priorities on the list of four health priorities for 1998 to 2008 (Personal communication, J. C. Mbanya, 2000). Most key informants reported that the cost of management of NCDs is so high that the fight for poverty alleviation may be jeopardised by increasing the priority rating. However, they acknowledge that many inadequacies exist, which are responsible for the poor care that is provided to patients at different levels of health care (Personal communication, J. C. Mbanya, 2000).

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1.0 0.9 0.8

Proportion

0.7 Cameroon Australia

0.6 0.5 0.4 0.3 0.2 0.1 0.0 0

250

500

750

1000

1250

1500

1750

2000

Days Since Seizure

Figure 3.1 A “time since seizure” graph showing the proportion of the Australian and Cameroon sample that had not had a seizure by a certain day.

In general, the perception of the condition in the society resulted in the internalisation of the sick role; an individual who was epileptic was considered to have a chronic illness and as result became chronically ill. Some of the specific symptoms reported included generalised itching and rash, sleepiness and dizziness. Most of the participants in the study complained of how much the sickness took out of them I never have energy because of this sickness

I have to be constantly bedridden because of this disease

I hardly go out, and when I do I can only go as far as the cock crows. [the short distance over which a cock’s crow can be heard] This perception of the chronic nature of epilepsy was reinforced by medical staff who advised participants, as routine management, to take things easy, forbidding exposure to the sun and advising against all strenuous exercise. The medical doctors who are treating this condition forbid us from having sexual intercourse or sexual partners. Even the doctors know that you cannot have sex with this disease.

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There were also a large number of food restrictions reported, recommended both by medical staff and traditional food taboos. All the participants reported that they were on some medication, although dosage and frequency depended on availability and affordability at any point in time. Some of the symptoms reported would be consistent with side effects of commonly used anti-epilepsy drugs (AEDs). However, because reported compliance was poor and participants had no knowledge of the possible side effects of their medications, there could be alternative reasons for the reported symptoms. Based on the “time since failure” analysis of their last seizure (Figure 3.1), their epilepsy would not be classified as being well controlled.

Gender factors in epilepsy in Cameroon There were eight single parents amongst the participants, all of whom were women. They were living with their parents either because their partners had left them after finding out they had epilepsy or because they knew about it but did not want to have to take responsibility for the children once they found out about the pregnancy. Women living with their families, particularly in the urban sample, were not allowed to go to the market unless accompanied by someone else - both to ensure that they were safe and to prevent the disclosure of their condition to the wider community. This had important implications for their ability to perform their domestic roles. For women in rural areas, one of the concerns was exposure to open fires for cooking, heat and light during the evenings. Major burns were reported to result from falling into open fires during seizures. Burns, for this reason are an important comorbidity of epilepsy, at least in Cameroon. (see Plate 3.11) Plate 3.11 Cooking facilities in rural Cameroon

Nineteen participants reported that they were abstaining from sexual intercourse on their doctors’ advice. Fourteen of these were male and they expressed concern about what this abstinence did for their self-esteem.

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Because of this illness, I cannot get work and I have to stay home all the time. Now the doctors also say that I cannot bed a woman. What is my purpose in this life with only relatives to look after me all the time?

Economic factors in epilepsy in Cameroon People with epilepsy in Cameroon are prohibited from approaching large bodies of water. Explanations for this taboo range from a belief that looking into water may cause their “heads to turn and cause a fit” to the possibility that they may drown if they had a seizure while in the water. Sixteen participants in the sample were from fishing communities along the Sanaga River and had been fishermen prior to their first seizure; their livelihood was removed by this taboo. In addition, those who lived in communities that required commuting over water became isolated from family and other supports (Plate 3.12). Other conditions that people with epilepsy were advised to avoid included being out in the sun and heights. Employment options were therefore greatly reduced because no manual job outdoors was possible, neither were common casual occupations that involved tree climbing such as fruit picking and palm wine tapping. Plate 3.12 River access – note the narrow footbridge in bottom left hand corner.

One participant had been in a white-collar job (with the presidential guard), but reported having problems with his memory. He eventually was relieved of his duties because of frequent time-off work and apparent vagueness. The people I worked with said I am becoming more and more disturbed so they sent me away. For the moment they only call me when there is a serious thing that needs my attention. When I get there I only look and tell them what to do and then I come back. I have no opportunity to advance in the job because of this thing.

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Forty out of the 44 participants reported that they could not afford to maintain a regular supply of medications†. In addition, the inability to work made supporting families very difficult. This illness has greatly paralyzed me, since I am doing nothing. I was driving but now I can’t do it anymore because I am sick and I have crises frequently, which hinders me from doing my work. My wife is no longer with me. You should know that with women, when one does not have money she cannot stay with you

Environmental factors in epilepsy in Cameroon A concern raised for participants in rural areas was the lack of availability of health services and medications. They had a long way to travel to the nearest available health centre and transportation was expensive. There were also occasions when prescribed medication was not available at the health centre dispensary or at a private chemist’s, or were not affordable. In spite of the reduced infrastructure in rural areas, five urban participants were keen to move there. They thought rural areas would be safer given the lower levels of road traffic, but they needed to trade this off against a perceived greater risk of exposure to witchcraft during vulnerable periods of a seizure.

Other social sequelae of epilepsy in Cameroon Epilepsy in Cameroon was reported as being highly stigmatised. Most participants faced high levels of discrimination, irrespective of environment, gender or socioeconomic status [“money does not stop stigma”]. Participants reported being expelled from school because their seizures were disruptive. Thirty out of the 44 participants had their education interrupted as a result of seizures. They reported that they were openly mocked and ridiculed even within their own homes, where they were not allowed to eat with the rest of the family. The neighbours treated me like a mad person. They would not let me talk to their children because they were afraid I would contaminate them. And when I was in the street they would point at me and say there is the mad man and shout at me to go away like a stray dog. There were also reports of family crises and rifts, with accusations of jealousy and the use of witchcraft to destroy successful or potentially successful members of the family by affliction with epilepsy. Although some treatment was available through primary health care centres, seizures were perceived to be caused by the supernatural. This perception was exacerbated by the irregular access to medication and the, subsequently, limited effect it had on the quality of life of people with epilepsy.

Epilepsy in Australia (Victoria) Support for individuals with epilepsy is provided primarily by the Epilepsy Foundation of Victoria (EPV), which has several offices in the Metropolitan and rural †

The project team raised money to obtain medications for some of the respondents.

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areas of Victoria. The EPV was founded by people living with epilepsy to promote awareness of and offer services to all people with epilepsy. Individuals with epilepsy may qualify for a disability pension and government subsidised rates on other goods and services. It is possible for people with epilepsy to hold a driver’s licence as long as they obtain medical clearance and comply with the National Guidelines for driving vehicles with epilepsy. These explain the seizure-free period depending on the nature of seizures, before driving is permitted. In general, most of the people with epilepsy who participated in the study in Australia were somewhat controlled on medication, and had access to and sought regular medical attention and to a lesser extent, social support networks. However, there were many complaints about the quality of care they received, with several under the impression that they were discriminated against because they went to their doctors very often and the doctors did not like to see them. I had a GP last year and I went there just about screaming for help, because I knew I had all the classic symptoms of depression and it was because I was on Oririx which just exacerbated the seizures, triggered them more often. I was made to feel like it was all my problem, that it was my fault. I saw another doctor who immediately referred me to a psychiatrist who also dealt with neurological disorders and I was admitted into hospital immediately. Based on the ComQol, the reported subjective quality of life was much lower than expected when compared both to the normal population and to the paraplegics in the study. One interpretation, based on other ComQol results, is that “homeostatic defeat” had occurred. This refers to the notion that there is a “set-point” around which subjective well-being sits. When events happen to improve or lower subjective wellbeing, homeostatic processes ensure that the person’s sense of well-being is, over a period of time, returned to the set-point [58]. This, it is suggested, is why someone with spinal cord injury may in the first months after the injury have a poor sense of subjective well-being, but that over a number of years, a sense of well-being returns. The ComQol results for the epilepsy participants, however, were so low that they suggested the homeostatic mechanisms had failed. An important health-related issue raised by the participants was the comorbidities associated with epilepsy. There were several descriptions of injuries and other complications that had arisen as a result of seizures. A 26-year-old described having a seizure while surfing which almost resulted in him drowning. He was rescued but needed reviving and ended up in hospital. The incident also signaled an increase in the severity and frequency of his seizures. Burns were also a comorbidity in the Australian study. These occurred during ironing, and cooking, but most commonly, when a seizure occurred during showers. There were reports of the use of a range of ‘recreational substances’ to cope with the depression and loss of self-esteem as a result of the condition.

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When I had regular fits, I started using different substances... to deal with the frustration and depression. A range of things made me either mellow or made me feel fantastic and unbeatable for 24 hours with no worry in the world and I would also get drunk to help me forget. A couple of times I came close to losing the plot, was having bad nightmares and almost topped myself because I could not deal with life and people kept nagging. What did they know? There were consistent reports of the side effects of AEDs being one of the worst problems of people with epilepsy, second only to stigmatisation. Participants complained about sleepiness (with two reports of insomnia), listlessness, hallucinations, dizziness, headaches and itching, and in general blamed the medication for aggression and anti-social behavior. The side effects of the medications were reported to affect general functioning within society and were responsible for an inability to study and obtain the desired level of education. Eight participants had had the ‘head operation’ (temporal lobectomy), which they reported as being reasonably successful in reducing the frequency and severity of seizures. I have tried so many different types of drugs I think I should be given a medal. One type they put me on made me really fat; I just ate and ate but the seizures seemed to be controlled so I did not worry too much. And then suddenly I had a huge big one [seizure] on the tiles at the back of the house and nearly cracked my head open, got concussion and was as sick as a dog as well on top of it. Now I am on something different again and I feel like I am dopey all the time. The continually changing medication means that I can be dull and listless on one day and high the next. This is a major thing to have to cope with; and try to live a normal life.

There have been many occasions in the last at least 5 years when I have sunk very low, I have been very depressed and the only thing that made me hang in there was this feeling that I should not let epilepsy take control of my life. I now firmly believe that it was the medication that made me depressed and not the epilepsy, which is a kind of strange distinction.

Gender factors in epilepsy in Australia Interview data suggested that the effects of epilepsy were different for women because the seizures were often linked to menstrual cycles, with some women describing a higher or lower frequency of seizures depending on where they were in their cycle. Three women reported that their seizures were best controlled when they were pregnant. However, the need to continue with medication made the planning of families difficult for some women, who were concerned about the effects of the medication on foetal development and on the interaction with the contraceptive pill. They also reported that medical practitioners they had seen advised them that they should not get pregnant because it was harder to maintain their drug levels and keep the seizures under control. Men in the sample complained about their loss of role and the lack of control over their lives, and their assumption of a “sick role” even when they were not having regular seizures.

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Both men and women were concerned about the effect of their seizures on intimate relationships. About half the sample was married and with the exception of one woman, had partners who were supportive of them. One woman reported that she had an abusive husband who thought she faked seizures for attention so she had to try as much as possible to: …keep my epilepsy in a little box and live my life around him because it is not fair he has to share it with me. One unmarried woman reported that she avoided sexual relationships altogether, because she did not feel like a sexual being and was not interested.

Economic factors in epilepsy in Australia There remain several professions that exclude individuals from employment on the basis of a medical history of epilepsy and this was a major concern, particularly with individuals who found they had to change professions following the diagnosis. I will soon be in my 40s and I am back to being an apprentice again. I used to be a roof tiler and I fell from a roof, which is how this whole thing started in the first place. I have now started doing clerical work.

I was working as a teacher until the principal found I had epilepsy. She then made every effort to get rid of me because she thought I would be exposing the children to emotional scarring if they witnessed me having a seizure. She was also one of those principals who was not keen on having children with any disabilities in the school

My previous job was as a receptionist at a doctor’s surgery. I lost the job soon after they found that I had epilepsy. You would have thought they would know better, or maybe I lost the job because they do. Some concern was also raised over other symptoms that were attributed to epilepsy, including a reduced ability to concentrate and increasing forgetfulness.2 These factors restricted the job options and consequently, economic potential. One participant reported an incident with a previous employer. He had fallen and the company had denied responsibility because they knew he had epilepsy and blamed the fall on a seizure rather than accept responsibility for the accident. Others reported that they had been passed over for promotion because they had had to take excess days off due to illness. The driving restriction for people with epilepsy was a major problem for mobility in general but created problems economically when particular jobs were contingent on the possession of a valid drivers’ licence.

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I have driven for years, trucks, courier vans and then this. I know that the rules say if I am okay for one year I can have it [driver’s licence] back but they make it really hard. What do the bureaucrats know, they make the rules and we have to follow them, they don’t understand what this means to our lives.

Environmental factors in epilepsy in Australia Economic and environmental issues were closely related. Rural participants talked about difficulties in obtaining employment because of the higher rates of unemployment. They spoke of how their epilepsy exacerbated the already difficult task of finding employment in rural areas because of people’s ignorance, their stereotyping of epilepsy, and difficulties moving around because of drivers’ license restrictions and the need to travel long distances. The inability to drive was one of the greatest concerns of most people with epilepsy but was particularly problematic in rural and remote communities. In the Melbourne metropolitan area, availability of public transport went some way to addressing this problem. However, through Photo Voice, a mother was able to describe the problems she had with travelling with her young toddler on the buses. Her child’s stroller carried detailed name and address labels. If the mother had a seizure, the child would be safer in the stroller than in her arms, and next of kin could be readily contacted. Because suburban buses have hooks for carrying stroller on the back of the buses, drivers were reluctant to let the mother keep her baby in the stroller on the bus (Plate 3.13). The negotiation with the driver required revealing her medical details within the hearing of other passengers on the bus. She was eventually successful in an application for special dispensation through the local government. For participants in rural areas however, their movements were very restricted unless they had carers they could rely on for transportation. Australia in general but particularly the “bush” is not suited for people without transport. Even people on the dole all the time can afford to get a banger and fix it up. I could probably afford a car but what good would it do me when I can’t drive it and instead I have to pick where I can go only if it is easy to get there. The issue of stigmatisation was also raised with the general comment that it was easier to be anonymous in large cities than in rural communities. In rural communities that are very small, everyone knows everyone else and for some things they are very supportive. But for epilepsy, it is still something that people have stereotypical expectations and they try to be careful around me to the point where I feel strongly like I don’t belong.

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Plate 3.13 It can be difficult travelling with small children in urban Australia

Other social sequelae of epilepsy in Australia All participants complained about the continuing stigmatisation in the community despite public campaigns to educate the general population about the condition and the ability of people with epilepsy to function “normally” when well controlled. There was a general feeling that once people found out about the condition they could “only see the disability and not the person”, and this had a profound impact on their functioning within their various roles even when their seizures were controlled. Most of the participants described losing friends, or finding it difficult to make friends and subsequently, there was a lot of loneliness and isolation. Paradoxically, participants also described choosing not to engage with others in the community. Even the bureaucracy does not understand epilepsy and tends to make blanket rules about issues like drinking, driving, insurance etc. Epilepsy education has not made the community more aware otherwise teachers and educators would be better than they are. And most important, if someone is having a seizure, you would find people helping them, instead they make cracks about them having a drug overdose reaction.

My mates always teased me about being vague and out of it. My teachers thought I was difficult and disruptive. My parents explained that I had complex partial seizures and so I would do odd things and have bizarre speech patterns. But I think just because I did not fall on the ground and foam at the mouth they thought we were just making excuses.

The hardest thing for me now is I am the only one amongst my friends who can’t drive

QUANTITATIVE ANALYSIS This section is sub-divided into four parts. The first part examines the paraplegia results. The second part examines the epilepsy results. The third part

46

compares the countries. The final part examines the relationship between the countries and the health conditions. The analyses revolve principally around the health-related quality of life (HRQoL) measures of the SF-12, and the subjective quality of life (SQoL) measures of the ComQoL. HRQoL is examined using the SF-12 mental health sub-scale (the MCS), the SF-12 physical health sub-scale (PCS), the SF-12 self-rated health item (question 1), the SF-12 self-rated pain item (question 5) and the SF-12 measure of physical and emotional problems (question 7). The scoring of the SF-12 MCS and PCS sub-scales followed the scoring for the US norms [46]. SQoL was examined using the mean satisfaction score from the seven ComQoL satisfaction items. An adjusted measure was also used which excluded the “satisfaction with health” question, but was still re-scaled 0 to 100. Similarly, the “satisfaction with health” item alone was examined. Analyses of the control, optimism, and self-esteem questions are not presented as a part of this report, but are being analysed for other publications. A “confidence interval” approach was often used in the analyses of these data. Parametric tests were also used (t-test and analysis of variance). In some cases a nonparametric test may have been more appropriate. However, where nonparametric equivalent tests were calculated, it made little difference to the interpretation of the data. Therefore, for ease of presentation, a single approach was used. Given the number of tests that were used in analysing these data, there was an increased chance of a Type I error, that is, there was an increased chance of falsely rejecting the null hypothesis. However, for two reasons no correction was made to protect against this. First, being an exploratory study, any inferential statistical tests were used largely as an adjunct to the descriptive analyses. Secondly, the outcome measures were often correlated, so the tests were not independent.

Paraplegia Cameroon – quantitative paraplegia data Table 3.1 shows means and 95% confidence intervals (95% CIs) on the HRQoL and SQoL measures. Median scores are included, where appropriate, as notes to the table.

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Table 3.1: Means and 95% confidence intervals of quality of life measures for participants with paraplegia from Cameroon. Measure MCS(1) PCS(2) General Health(3) Pain(4) Physical/Emotional(5) ComQoL(6) ComQoL – Without Health(7) ComQoL—Health(8) (1) (2) (3) (4) (5) (6) (7) (8)

Mean

n

Lower 95% CI

Upper 95% CI

36.2 29.2 1.6 3.75 4.1 45.6 48.5

34 34 40 40 40 39 39

33.9 27.8 1.3 3.4 3.8 41.3 44.2

38.6 30.6 1.8 4.1 4.4 49.8 52.8

25.6

40

18.7

32.5

The SF-12 mental sub-scale score The SF-12 physical sub-scale score SF-12 self rated health question – the median rating was 1 (“poor”) SF-12 self-rated pain question – median score was 4 (“quite a bit”) SF-12 self-rated physical/emotional problems – median score was 4 (“most of the time”) The mean score of the ComQoL satisfaction questions normalised to fall between 0 and 100 The mean score of the ComQoL satisfaction questions excluding the health question normalised to fall between 0 and 100 The ComQoL question about satisfaction with health normalised to fall between 0 and 100 – median score on the original scale was 2 (“unhappy”)

The MCS and PCS scores were well below the US population norms. This is necessarily so for the physical scale because the participants would answer “no:” to questions about walking and climbing. However, the low MCS scores suggest a number of possibilities including either, that as a population, the people in Cameroon would score well below the US population, or the sub-population of people with paraplegia suffer significant mental and emotional problems. Without population norms for Cameroon there is considerable uncertainty in the interpretation. It is interesting to note however, that they self-rated their general health as lying on average between “poor” and “fair”. Given the general assumption that spinal cord injuries are often associated with a lack of sensation, it is also worth noting that most participants reported that pain interfered with their lives “quite a bit” (40%) or “extremely” (24%). The mean SQoL score as measured by the ComQoL was 45.6. Cummins reported that a score of between 60 and 80 could be expected in a developing country in almost any population [61]. The 95% confidence interval surrounding the estimated SQoL score suggests that on average their quality of life falls well below that of the population mean, even if the population mean is at the low end of expectation – 60. This would further suggest a failure of homeostatic control [58]. One possible explanation for the very low SQoL score is that the participants’ satisfaction with their health dragged down the overall mean. When SQoL is reestimated, excluding the “satisfaction with health” item the 95% confidence interval still excludes 60 (44.2 to 52.8). There was, however, a significant difference between SQoL estimated with the health item and without the health item included (difference between the means = 2.96, 95% CI: 1.93 to 3.99). Gender, environmental, and socioeconomic status (SES) differences were examined with t-tests. No differences were found between the male and female

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participants with paraplegia on any of the HRQoL of SQoL measures. A significant difference was found between urban and rural dwellers on the SF-12 physical health sub-scale. The mean urban PCS score was 30.9 and the mean rural score was 27 (difference between means = 3.95, 95% CI: 1.52 to 6.38). This indicated that the rural dwellers were significantly worse off, with respect to their physical health, than were urban dwellers. A significant difference was also found on the SF-12 physical sub-scale between participants categorised as coming from high or low SES. The mean PCS score was 27.9 for people from low SES and 30.6 for people from high SES (difference between means = 2.7, 95% CI: 0.10 to 5.37). This indicated that participants from a high SES background were significantly better off, with respect to their physical health, than were participants from a low SES background. An analysis of variance with SES and environment as fixed effects and PCS as the dependent measure suggested that environment was a significant predictor of PCS and that SES was not. The interaction effect of environment by SES “approached” significance (p=.096). Environment alone accounts for around 23% of the variance of the PCS scores.

Australia - quantitative paraplegia data The approach taken with the Australian data was almost identical to that taken with the Cameroon data. Table 3.2 shows means and 95% CIs associated with the quality of life measures for the participants with paraplegia. One of the striking features of the following table is that only 9 (25%) of the participants with paraplegia completed enough of the SF-12 to calculate the mental health sub-scale (MCS) and only 13 (36%) completed enough of the SF-12 to calculate the physical health sub-scale (PCS). This response rate was so low that no further analyses were carried out with these scales. Fortunately, three of the SF-12 items of interest were well completed. The median response for self-rated health was “very good”, there was little reporting of pain interfering with life (the median response was “a little bit”), and the median response for physical or emotional problems interfering with life was “a little of the time”. Twentythree (64%) participants reported that they were “mostly satisfied”, “pleased”, or “delighted” with their health when answering the health-related item in the ComQoL. Mean subjective quality of life as measured by the ComQoL was 73.7. Cummins reported that in “western countries” one could expect the population mean to sit between 70 and 80. In Australia, this score is around 75 [47]. When the ComQoL score was recalculated with the satisfaction with health item excluded it rose slightly to 75.2. Although both these results were consistent with the Australian population norm, the inclusion of the satisfaction with health item reduced the ComQoL score significantly (mean difference=1.48, 95% CI: 0.69 to 2.28).

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Table 3.2: Means and 95% confidence intervals of quality of life measures for participants with paraplegia from Australia. Measure MCS(1) PCS(2) General Health(3) Pain(4) Physical/Emotional(5) ComQoL(6) ComQoL – No Health(7) ComQoL—Health(8) (1) (2) (3) (4) (5) (6) (7) (8)

Mean

n

Lower 95% CI

Upper 95% CI

35.4 31.4 3.5 2.3 2.3 73.7 75.2

9 13 35 36 36 36 36

28.97 29.41 3.10 1.79 1.84 69.1 70.8

41.81 33.37 3.93 2.71 2.78 78.3 79.6

58.6

36

50.79

66.42

The SF-12 mental sub-scale score The SF-12 physical sub-scale score SF-12 self rated health question – the median rating was 4 (“very good”) SF-12 self-rated pain question – median score was 2 (“a little bit”) SF-12 self-rated physical/emotional problems – median score was 2 (“a little of the time”) The mean score of the ComQoL satisfaction questions normalised to fall between 0 and 100 The mean score of the ComQoL satisfaction questions excluding the health question normalised to fall between 0 and 100 The ComQoL question about satisfaction with health normalised to fall between 0 and 100 – median score on the original scale was 4 (“mostly satisfied”)

Gender, environmental, and SES differences were examined with t-tests. No differences were found between urban and rural dwellers with paraplegia on any of the measures. Similarly, no differences were found between the male and female participants. However, on the pain item from the SF-12, the difference between the mean male (mean=1.8; 95% CI: 1.30 to 2.38) and female (mean=2.7; 95% CI: 1.94 to 3.47) self reported pain scores “approached” significance (t=1.98; df=33 p=.056) with female participants reporting higher levels of pain. There were significant differences between high and low SES participants on the self reported pain item from the sf-12, the ComQoL score, the ComQoL score recalculated without the satisfaction with health item, and the satisfaction with health item alone (Table 3.3). On all tests reported, the participants with paraplegia from low SES scored worse than those from a high SES. Their ComQoL scores were 13% of the scalemaximum lower than those from high SES. This was the case with or without the satisfaction with health item included in the scoring.

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Table 3.3: Differences between means on quality of life scores for participants with paraplegia from high and low SES backgrounds in Australia. Measure Pain(1) ComQoL(2) ComQoL – No Health(3) ComQoL—Health(4) (1) (2) (3) (4)

Mean Difference

t

df

p

1.01 -13.5 -13.16

2.26 -3.4 -3.52

31 31 31

.0307 .0019 .0014

-15.44

-2.22

31

.0335

SF-12 self rated pain question The mean score of the ComQoL satisfaction questions normalised to fall between 0 and 100 The mean score of the ComQoL satisfaction questions excluding the health question normalised to fall between 0 and 100 The ComQoL question about satisfaction with health normalised to fall between 0 and 100

Epilepsy It was shown in Figure 3.1 that the pattern of the time since the last seizure was very different between the Australian and the Cameroon participants, with the Australians appearing to have much better controlled epilepsy than the Cameroonians. The mean time since the last seizure for Australian participants was 266.3 days, while it was only 50.9 days for participants from Cameroon. A question, therefore, arises about whether any differences that may be observed between Australia and Cameroon on the subjective and health-related quality of life data would be attributable to the countryfactors or to the clinical differences in the presentation of the epilepsy. The epilepsy data were split into participants who had a recent seizure and participants who had not. The split-point was the median time since the last seizure, which happened to be 30 days for both the Cameroon and Australian participants. A series of analyses of variance were used to test whether observed differences in subjective and health-related quality of life were significantly associated with country, time since last seizure, or the interaction effect of country by time since last seizure. No significant difference was found between participants with more or less recent seizures on any of SQoL or HRQoL measures. The time since last seizure was, therefore, excluded from any further analyses.

Cameroon – quantitative epilepsy data Table 3.4 shows the means and 95% CIs for the quality of life measures completed by the Cameroon participants with epilepsy. The interpretation of these results is very similar to the interpretation of the paraplegia results for Cameroon. The MCS and PCS score were well below the US population norms. Self-rated general health lay between “poor” and “fair”—with “poor” being the median response. On average, the participants reported moderate pain. They also reported on average that physical or emotional problems affected their lives “some of the time”.

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Table 3.4: Means and 95% confidence intervals of quality of life measures for participants with epilepsy from Cameroon. Measure MCS(1) PCS(2) General Health(3) Pain(4) Physical/Emotional(5) ComQoL(6) ComQoL – No Health(7) ComQoL—Health(8) (1) (2) (3) (4) (5) (6) (7) (8)

Mean

n

Lower 95% CI

Upper 95% CI

33.1 29 1.5 3.1 2.8 54.2 57.8

26 28 42 42 42 41 41

31.00 27.54 1.26 2.73 2.41 49.15 52.31

35.13 30.37 1.78 3.46 3.16 59.28 63.31

32.6

42

25.05

40.19

The SF-12 mental sub-scale score The SF-12 physical sub-scale score SF-12 self rated health question – the median rating was 1 (“poor”) SF-12 self-rated pain question – median score was 3 (“moderately”) SF-12 self-rated physical/emotional problems – median score was 3 (“some of the time”) The mean score of the ComQoL satisfaction questions normalised to fall between 0 and 100 The mean score of the ComQoL satisfaction questions excluding the health question normalised to fall between 0 and 100 The ComQoL question about satisfaction with health normalised to fall between 0 and 100 – median score on the original scale was 2 (“unhappy”)

Mean subjective quality of life as measured by the ComQoL was 54.2 (95% CI: 49.15 to 59.28). The 95% confidence interval excluded the lower limit of the population norm (60) that could be expected in a developing country. When subjective quality of life is recalculated, excluding the “satisfaction with health” item, the 95% confidence interval includes 60 (52.31.2 to 62.31). Gender, environmental, and SES differences were examined with t-tests. No differences were found between the male and female, or high and low SES participants with epilepsy on any of the HRQoL of SQoL measures. There was a significant difference between the level of physical and emotional problems (from the SF-12) reported by the participants with epilepsy in urban and rural Cameroon. Those in urban Cameroon reported that physical or emotional problems interfered with their work less than those in rural Cameroon (mean difference=0.83; 95% CI: 0.121 to 1.542).

Australia - quantitative epilepsy data The approach taken here is principally the same as the approach taken with the Cameroon data. Less detail, however, is provided as the results become more repetitious. Table 3.5 shows the means and 95% CIs for the quality of life measures used with the Cameroon participants with epilepsy.

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Table 3.5: Means and 95% confidence intervals of quality of life measures for participants with epilepsy from Australia. Measure MCS(1) PCS(2) General Health(3) Pain(4) Physical/Emotional(5) ComQoL(6) ComQoL – No Health(7) ComQoL—Health(8) (1) (2) (3) (4) (5) (6) (7) (8)

Mean

n

Lower 95% CI

Upper 95% CI

29.7 28.4 3.1 2.0 2.4 66.4 67.5

15 13 34 34 34 34 34

26.28 25.9 2.72 1.62 1.99 61.52 62.4

33.08 30.83 3.46 2.44 2.78 71.25 72.55

59.9

34

51.9

67.81

The SF-12 mental sub-scale score The SF-12 physical sub-scale score SF-12 self rated health question – the median rating was 3 (“good”) SF-12 self-rated pain question – median score was 2 (“a little bit”) SF-12 self-rated physical/emotional problems – median score was 2 (“a little of the time”) The mean score of the ComQoL satisfaction questions normalised to fall between 0 and 100 The mean score of the ComQoL satisfaction questions excluding the health question normalised to fall between 0 and 100 The ComQoL question about satisfaction with health normalised to fall between 0 and 100 – median score on the original scale was 5 (“mostly satisfied”)

As with the Australian participants with paraplegia, the participants with epilepsy had high levels of missing data making the calculation of the mental health (MCS) and physical health (PCS) sub-scales of the SF-12 possible only for a relatively small percentage of the participants. Fifteen (44%) of the participants completed items for the MCS and 13 (38%) completed enough of the SF-12 to calculate the PCS. This response rate was so low that no further analyses were carried out with these scales. The SF-12 items dealing with self-rated health, pain, and physical and emotional problems were well completed. The median response to the self-rated health item was “good” with 44% (14) of the respondents rating their health as “very good” or “excellent”. Sixteen (47%) reported that pain did not interfere at all with their normal work and 11 (32%) reported that it interfered “moderately” or “quite a bit” with their normal work. Average subjective quality of life as measured by the ComQoL was 66.4 (95% CI:61.52 to 71.52). This was not significantly lower than the lower normative score in “western” populations of 70 [61] but it is lower than the Australian norm (75). When the ComQoL score was recalculated without the health item it rose slightly to 67.5. These scores were not significantly different (mean difference=1.09, 95% CI: -0.03 to 2.20). Gender, environmental and SES differences were examined with t-tests. No differences were found between the males and females, or urban and rural participants with epilepsy on any of the HRQoL or SQoL measures. There was a significant difference in satisfaction with health reported by the participants from different SES with people from high SES reporting more satisfaction with their health (mean difference=16.8; 95% CI: 1.04 to 32.64).

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COMPARING THE COUNTRIES In this section, an analytic approach is taken toward the quantitative data. The focus is on the degree to which the factors (Country, Disease, Gender, SES, and Environment) can explain the quality of life measures between the countries.

Paraplegia – country comparisons It is quite clear, even without the formal calculation of inferential statistics, that the variation between Cameroon and Australia on the health-related and subjective quality of life measures was substantial. Figure 3.2 illustrates this with the self-rated health question from the SF-12 completed by people with paraplegia in Cameroon and Australia.

0.6 Australia Cameroon

Proportion of Sample

0.5

0.4

0.3

0.2

0.1

0.0 Poor

Fair

Good

Very Good Goof

Excellent Excellenv

Self Rated Health

Fig 3.2 Self-rated health of participants with paraplegia living in Cameroon and Australia

Analyses of variance were conducted using country, gender, SES, and environment as fixed factors. The self-rated health, pain, and physical and emotional problems items from the SF-12 and the mean satisfaction score from the ComQoL were the dependent measures. All possible two-way interaction effects were included in the models but no higher order interactions effects were included. The pattern was very similar in all cases. ‘Country’ overwhelming accounted for most of the variance in the dependent measures. There were either no other significant effects, or it was SES. In one instance, there was a significant interaction effect between country and gender.

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Epilepsy – country comparisons A similar approach was taken with the data from the participants with epilepsy. Analyses of variance were conducted using country, gender, SES, and environment as fixed factors. The self-rated health, pain, and physical and emotion problems items from the SF-12 and the mean satisfaction score from the ComQoL were the dependent measures. All possible two-way interaction effects were included in the models but no higher order interactions effects were included. ‘Country’ was the only significant effect, in all but one analysis. In the latter analysis there was a significant interaction effect of country by environment on the physical and emotional problems item from the SF-12. This effect explained around 4% of the variance.

Country by Disease Analyses of variance were conducted that included the health condition as well as country, gender, SES, and environment as fixed factors. The self-rated health, pain, and physical or emotional problems items from the SF-12 and the mean satisfaction score from the ComQoL were the dependent measures. The second order interaction effect of country by health condition was also included in the model. There was no particularly consistent pattern in the results. Country was the only factor significantly associated with self-rated health. Country and health condition were significantly associated with self-rated pain. Country, health condition and the interaction effect were significantly associated with physical or emotional problems items from the SF-12. Country alone and the country by health condition interaction effect were significantly associated with the mean subjective quality of life of the ComQoL. A graph of this final relationship is shown in Figure 3.3. This figure suggests that in terms of subjective quality of life, people with paraplegia may be better off in Australia than people with epilepsy, but people with epilepsy may be better off than people with paraplegia in Cameroon. This result is in some ways counter-intuitive and possible reasons for this are discussed in the following chapter. The next chapter forms the main discussion of the report. Data are integrated with theory, and the DALY (specifically the global severity weight) is scrutinised in the light of the two.

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Subjective Quality of Life Score

90

80

70

60

50

40

Epilepsy Paraplegia

30 Australia Australic

Cameroon

Country

Figure 3.3 Mean reported satisfaction from the ComQoL for people with paraplegia and epilepsy living in Cameroon and Australia – 95% confidence intervals are shown

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NOTES 1

In general, paraplegia is reported to be a terminal condition in Cameroon because it is expected that death would occur from generalised infections resulting from pressure sores. 2 It should be noted that these symptoms are also consistent with commonly reported side-effects of some anti-epileptics such as tiredness, confusion, depression and mood swings.

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CHAPTER 4 DISCUSSION In this chapter, we examine the limitations of the study design, the broad thrust of the results and their implications for the measurement of the burden of disease. We start by considering the limitations.

Limitations of the study design The research process was carefully documented to minimise potential problems that could arise given that data collection was conducted by two teams in different countries. The following limitations, however, need to be discussed because of the bearing they have on the interpretation of the results. •

Were “like” health conditions compared;

•

Sampling;

•

The cross-cultural validity of the instruments; and

•

The use of self-report data These are discussed seriatim.

Comparing “like” health conditions In the determination of the burden of disease, the assumptions on which the DALY is based require comparisons of “like with like” [2] (p.6). In the choice of conditions for the study and in the recruitment of participants, therefore, we attempted to recruit participants with similar clinical manifestations of the chosen conditions. The first problem that arose was with the identification of the conditions to be used for the study. The original proposal for the study suggested a number of possible conditions to examine including hearing impairment, diabetes, rheumatoid arthritis and blindness. Although hearing impairment for instance would have been possible to study in Australia there were a number of problems in Cameroon. The health and education services in Cameroon have a very limited capacity to assess and support children with congenital or early-acquired hearing loss. Thus, in the absence of any other impairments, most children born with total hearing loss are also unable to communicate through Sign or other language, are often ostracised and to all intents and purposes are treated as intellectually disabled. In the longer term, mental health problems due to social isolation are some of the possible sequelae. Rheumatoid arthritis was also not a feasible condition to study because of the difficulties of diagnosis in both urban and rural areas in Cameroon. Similar problems occurred with diabetes. Because the systems for the early detection of diabetes are better developed in Australia, an identified population of Australian diabetics is likely to have a less severe clinical presentation than a population of identified Cameroonian diabetics. The process of selecting the particular conditions for the study highlighted the underlying problem of what appeared in the first instance to be a relatively simple task – identify people with a health condition with similar clinical manifestation in a developed and developing country.

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In selecting participants with epilepsy for this study, one control was to include only those participants on medication – this would ensure a more similar clinical manifestation of the condition. A comparison of treated epilepsy (Australia) with untreated epilepsy (Cameroon) would not have informed the question about social milieu at all. Fortunately, anti-epileptic drugs (AEDs) are available in Cameroon [62], although anecdotal evidence also suggests that the compliance is lower than expected. Notwithstanding that, the AEDs in Cameroon are relatively more expensive than in Australia, where they are publicly subsidised as a part of the national health system (the Pharmaceutical Benefits Scheme). Thus, the Australian participants have more regular access to medication. Furthermore, even if the two groups were equally compliant about taking their medication, because of the costs, the Cameroon sample were taking AEDs that are (a) an older generation of AEDs with more pronounced side-effects and (b) manufactured with poorer quality controls making the dosage less certain.1 Thus, ensuring the clinical comparability of the epilepsy was not simple. With other conditions, treatments varied substantially across and within countries depending on financial resources, and the availability of treatment. This made it difficult in an international study to ensure that “like” was being compared with “like”, even with a priori inclusion and exclusion criteria for participation, such as “treatment”. In the study of The Burden of Disease and Injury in Australia [10], it was noted that, In order to make a consistent and meaningful estimate of YLD for a condition, it is crucial to clearly [sic] define the condition under consideration in terms of case or episode, and severity level or disease stage [clinical manifestation]. It is then necessary to ensure that the disability [severity] weight and the population incidence/prevalence data relate to the same case definition. The most difficult step in estimating YLD for most diseases is matching existing population data to the … [clinical manifestation] for which the weights are available. Getting this wrong can result in substantial errors in the YLD estimate. (p.21) (Authors’ italics)

Our data and the above quote indicate that the problem of obtaining “like” cases in an international study is probably of less threat to the validity of the present study than it is to the general efforts at estimating the global burden of disease. Indeed, because of the importance placed on the collection of high quality qualitative data, differences in the clinical manifestation between countries merely served to illustrate the complexity and the range of health outcomes experienced by people with the particular health condition. In contrast the global burden of disease studies assume homogeneity in the clinical manifestation of the “treated” and “untreated” health conditions between countries.

Sampling Another potential limitation of the study was the sampling. The sampling strategy used for the study was purposive but opportunistic with snowballing. The participants were self-selected and may have opted to participate for a variety of reasons from genuine interest and a desire to help, through to a desire to advocate a personal agenda. There are obvious problems with this but there was necessarily a trade off between a statistically ideal sampling method and what was feasible within the constraints of time and resources. There are some evident biases, particularly in the choice of Australians with paraplegia both in the timing of the study (see Chapter 3) and the recruitments from the Wheel Chair Sports Association. Data from less well off participants were obtained as well which does provide some balance to the data and this is an issue that could and should be addressed in a larger study.

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Furthermore, the strengths of the data lie not only on the quantitative measures which are based on the conventional public health methodology, but particularly on the qualitative data which was crucial in obtaining rich and detailed descriptions of living with the conditions. Although the data are not exhaustive in capturing the full range of experiences, they still provided us with a greater insight into the “average handicap” for the particular context.

Cross cultural validity There were some problems with cross cultural interpretations of the various items on the standardised instruments which raised questions about the validity of the scores on the various scales. Nonetheless, the magnitude of some of the differences were so great, that it would seem more plausible to seek an explanation not just in the interpretation of the scale items, but also in the conditions of the participants lives. It was here that the qualitative data was of particular importance. By examining the quantitative data in conjunction with the qualitative data, it became much clearer why participants in Cameroon could be scoring so much lower than Australian participants on a number of scales. Some of the explanation may lie in the cultural interpretation, but some of it would also appear to lie in the context in which the lives were lived. Pre-testing the instruments provided the opportunity to amend some of the questions where necessary. In addition, the qualitative methodology was essential in highlighting and addressing areas where participants had problems understanding the questions. Several participants in Cameroon who engaged in the Photo Voice exercise asked the researchers to take the photographs. This was probably due to their inexperience with the use of cameras. However, the purpose of the methodology was to ensure that participants generated the images that reflected the impact of the health condition on their lives. The delegation of the activity to the research assistants meant that the images may better portray the research assistants’ interpretations of the life of a participant. Consequently, the photographs were used mainly as a tool for further interviewing and not analysed contextually for the visual representations in themselves. There were some problems with the Photo Voice method with the participants in Australia; the cameras obtained were of poor quality, which in turn affected the quality of the photographs. The participants described and explained the photographs they had taken and so as a tool for interviewing, the method was somewhat effective. However, this is a practical consideration for the use of the method.

Self-report data The use of self report data in general has raised some real and legitimate concern on the part of the authors of the DALY (e.g., [2]). They highlight the fact that individuals’ perceptions and reporting of their own health status may not coincide with their actual health status [25] It is not argued here that self-report data are without bias, but that interviewing people about their daily experience of their health condition, including functional and emotional aspects, provides important insights into the relationship between context and severity. Indeed, relatively little was asked about people’s health status. The general focus was on the relationship with the world around them.

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Notwithstanding the limitations presented here, the data appear to be robust and the general message from the data is clear. Context can have a large and significant impact on the severity of a health condition. Discussion on the self reported subjective quality of life of people with paraplegia and people with epilepsy are discussed elsewhere2. In the next section we examine specifically the broad implications of the results for the measurement of the burden of disease, particularly as they relate to the DALY are discussed.

Implications Much of the difference between the severity of paraplegia in Cameroon and Australia appears to relate to the level of development between the two countries. There are clear differences in the macro (e.g., paved roads and user-friendly housing) and micro (e.g., wheelchairs) infrastructure that improves the mobility of the population with paraplegia. Thus, one might expect a gradient of severity, with severity lessening as the development of a country improves. The effect of the development gradient has implications for the estimates of burden of disease that have been made at the country and regional level. In the less developed countries (where severity is worse than that indicated by the global average handicap) the burden of disease will be greater than estimated, and in the more developed countries, it will be less than estimated. There is the potential for health inequalities to be exacerbated and the chronic undercounting of severity in less developed countries could then feed into poor decisions about health priorities. It would probably not, however, be enough to build a “development adjustment factor” into the DALY. Although a development gradient may explain the difference between the severity of epilepsy in Cameroon and Australia, the magnitude of the gradient is not the same as the paraplegia gradient. The reversal in the rank order of the quality of life measures between the epilepsy and paraplegia in Cameroon and Australia suggests that development is not the only factor, and other factors including more culture-bound influences were at play. This finding cannot be regarded as isolated to paraplegia and epilepsy, and is relevant to other health conditions as well. So, does an indicator have to account for context and if it does, is it equitable? We examine these questions by revisiting some of the fundamental concepts of the DALY and relating them to debates about the importance of context in the measurement o f the burden of disease. We review the definitions of health outcomes, specifically disability; we examine egalitarianism and equity in the DALY and we highlight briefly some considerations for resource allocation.

Health is context independent – or no-context is the right context Since the conception of the DALY there has been a shift in thinking from no consideration of context (disability) through to a partial consideration of context (average handicap). The literature on this subject can be somewhat confusing and a cursory reading that focuses on one time-period could lead to apparently contradictory interpretations. This would occur particularly when reading criticisms in conjunction with the original works. For example, one critical analysis of the DALY [27] commenting on the 1994 presentation of the ideas was submitted for publication in 1995 [3]. It was accepted for publication in December 1996, after a new evolution DALY thinking had already been published [2]; but it was not actually published until 1997, in the same journal issue in which a third evolution of DALY thinking was published [25].

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Defining health outcome In the World Development Report 1993 (WDR), where the DALY made its debut, the severity of a condition was discussed in terms of “disability” [1] (p.26).3 In 1994 [3], when describing the technical basis for the DALY, again the discussion was in terms of disability (p.11), with specific reference made to the definition of disability in the International Classification of Impairments, Disabilities, and Handicaps [26]. Importantly, “handicap” was rejected as a definition for the purposes of evaluating the severity of a health condition. This notwithstanding, it was clear that handicap was being considered more than it had been before ([2] see note 33 on page 73 in reference to the text on page 34) and in Rethinking the DALY [2] the pendulum had definitely swung toward “handicap”. Unfortunately, even within this latter paper the discussion borrowed from earlier work that was restricted to disability alone [2]: …[T]he interest is in quantifying the burden from a condition given average conditions of individuals and social responses. (p. 9) In the terminology of the International Classification, Impairments, Disabilities, and Handicaps, I have chosen to measure disability not handicap. (p.33) Handicap is an attractive concept because it focuses on the particular social context of the individual. … In many cases, however, allocating resources to avert handicap as opposed to disability could exacerbate inequalities….. To avoid the obvious problems with such an approach, one must focus on disability rather than handicap. (p. 33) Having stated this, the actual approach … has been to use a construct somewhere between disability and handicap. More precisely … the concept is best described as average level of handicap. (p. 33-34) In this exercise [the person-tradeoff] the distinction between disability and handicap is somewhat blurred. Nevertheless, for the calculation of the DisabilityAdjusted Life Years, we do not want to factor in very specific circumstances where a disability may or may not lead to a handicap because of a particular social condition. Rather assessment should be based on average social conditions. (p93-94).

This confusion, however, is completely resolved in a paper the following year – 1997 [25]: [P]articipants … were asked to evaluate the average individual with the condition described taking into account the average social response or milieu for the world. (p.713) In the terminology of [ICIDH]…we have instructed individuals in the various disability [severity] weight exercises to evaluate the average handicap from a disability. (p.713 – footnote) [The DALY] is a measure of those functioning achievements (or lack of them) that can be specified due to illness (p.724 – footnote)

The move from disability to average handicap in some ways “opened the door” for the critique of the DALY presented here. For some authors, this progression from disability to average handicap has been unsatisfying. There has been some suggestion that summary measures of population health should focus on health dimensions such as impairments and activity limitations that lie “within the skin” or are intrinsic to the person. Furthermore, the suggestion is that one should ignore the interaction between the environmental and social contexts and the individual [10, 64, 65].4 By removing any consideration of the interaction between the individual and the social and environmental

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contexts, the problem with disaggregating the burden of disease data by region, country, sex, etc., would be solved. One would no longer need to use an average social milieu in the estimation of the global severity weight, because the social context is gone; and because of that, there would be no need to show that the social milieu was similar in all places.

Disadvantages with the loss of context This solution would be an illusion. There are problems with the loss of the context in the evaluation of the severity of a health condition. If the intention of the exercise was to provide a measure of the burden of disease in populations, then it is important that one measures the burden, as much as possible, as it truly occurs, not in an arbitrarily distorted reality. The experience of a health condition is an interaction between a person and their social, cultural, and environmental contexts – as the data presented in Chapter 3 amply demonstrate. Indeed, the edges between functional limitations that are intrinsic and extrinsic begin to blur as one examines those limitations in different contexts. In Australia people with paraplegia have wheelchairs; for some of them the wheelchairs are motorised and the loss of mobility is reduced. In Cameroon many do not have wheelchairs, and their mobility is severely restricted. Wheelchairs do not lie within the skin, so it is the interaction between the individual and things extrinsic (i.e. the wheelchair) that improves the mobility. When this ‘blur’ was argued in academic fora, there was some disagreement. The argument against was that the provision of a wheelchair is essentially treatment and therefore relates to the intrinsic nature of the condition. Even if this point were conceded, however, moving the ‘treatment’ one step further away from “within the skin” is not hard. Wheelchairs alone are of no use unless the environment is designed to cater for wheelchairs. Australia has building codes and laws covering equity of access for people with disabilities that mean that wheelchairs are a useful intervention to improve the mobility of people with paraplegia. More importantly, these rules have led to an improvement in access to buildings and public spaces. In Cameroon, the basic infrastructure to support the free movement of a wheelchair is not available. So, the provision of a wheelchair in one context would not have the same “treatment effect” that it would have in another context. Even something as apparently individual and “within the skin” as pain is not free from the interaction with the society. Stressful environments (including social and cultural environments) are known to increase levels of pain in people with chronic pain [66, 67]. Reducing stress not only improves the ability to cope with a certain level of pain, it also decreases the pain itself.

Analysing the social determinants It has been said of the DALY that it is too biomedical in its focus and that it does not permit an analysis of the social determinants of the burden of disease [27-29, 32]. This criticism has been rejected by Mathers [64] who attributed the criticisms of the DALYs ‘excessive’ biomedical focus to a conceptual fallacy (p.21), arguing that the DALY was actually very useful in the analysis of the social determinants of the burden of disease. Critics of the DALY, he argued, had failed to realise that an outcome measure of population health need not include all the important determinants of that outcome in order to be used in analyses that look at the relationship between the determinants and the outcome measure. Indeed, it has become a common refrain of DALY proponents that it can be used to examine the social determinants of the burden of disease (e.g., [2, 10]).

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We believe, however, that Mathers’ argument missed at least one part of the point of the critics. In order to detect a relationship between two measures they must covary (e.g., as one changes, so too does the other). If a single global severity weight is used for each health condition, it is by definition fixed in all contexts and cannot co-vary with any factors. Any analysis, therefore, of effects of context on the severity of a health condition (if a single global severity weight is used), will show no association. There has been a failure to recognise that two different types of social determinants of the burden of disease exist. There are (a) the social determinants of a health condition and (b) the social determinants of the severity of the health condition. For instance, one social determinant of paraplegia is the strength of the regulatory environment, particularly as it relates to industrial, building and road safety. A social determinant of the severity of the condition, however, is the level of infrastructure available that makes living with paraplegia easier. Mathers recognised the former type of social determinants, but not the latter. HIV/AIDS is an example of how the two types of social determinants differ from, and yet can be related to, each other. The level of stigmatisation in some societies negatively affects the lives of those who are HIV positive and thus increases the severity of the condition. Those living in more stigmatising societies warrant a higher severity weight than those living in less stigmatising societies. Stigmatisation also drives the disease underground and makes it harder to deliver health promotion/safe-sex campaigns, which in turn increases the population’s risk of infection. An analysis of the social determinants of disease would reveal both. In ignoring the social determinants of the severity of the health condition, there is a critical opportunity lost to avert the burden of disease. The use of the global severity weight hinders the accurate measurement of population health, and therefore interferes with the possibility of sound management.

Context and egalitarianism are incompatible. In spite of arguments that the DALY explicitly needs to exclude context in order to enhance its strongly egalitarian flavour [2], inequities in the DALY associated with the lack of context have been raised, notably by Anand and Hanson [27, 28]. The following counterexample was used to illustrate why one would not want to include contextual factors (in this case socioeconomic factors) in the construction of the DALY. Imagine a situation where two patients arrive at an [hospital] emergency room both in a coma from meningitis, but there is only enough antibiotic to treat one of them. The two patients are totally identical in every respect except that one is rich and the other is poor. The welfare maximisers might choose to treat the rich patient, while others … would choose to treat the poor patient. I [C.J.L. Murray] argue that … we should be completely indifferent to one over the other. [2] (p.7)

Essentially, this was an argument for the equality of resource allocation. The fact that the health condition in the example included coma is instructive; it suggests that two elements of the DALY have been conflated. Recall that in the calculation of the DALY, the YLD component is a function of (a) social preferences (e.g., age sex and time discounting), and (b) the severity of the health condition. In the meningitis example, it is clear that contextual information about the wealth of the patients should be ignored because if it were not, there would be an explicit social preference for the life of, say, a rich person over a poor person. However, because the two patients were comatose, they could not interact with their environment. Therefore contextual factors such as social, cultural and environmental factors could not make the

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state more or less severe. Thus contrary to the general tenor of the argument, it is not a general example about the exclusion of context in evaluating the burden of disease. Instead it presents an argument about the exclusion of specific social preferences. This, nonetheless, leaves open the possibility of having contextual information influence judgements about the severity of a health condition. It would be difficult to obtain consensus on the inclusion of a social preference in the DALY reflecting the value of the life of a rich person over that of a poor person. On the basis of the data collected here, it would seem to be perfectly reasonable to expect broad consensus about the fact that contextual factors affect the severity of a condition, e.g., that a rich person is more able to obtain the goods and services that could make a health condition less severe than a poor person. The equality in the DALY that is argued in the meningitis example above is sought in the domain of resource allocation and not in the measurement of the burden of disease. Everyone with a particular health condition without regard to the context of that health condition is to be treated equally. Equality, however, can occur over any number of domains, and equality in one domain will often result in inequality in another domain [68]. An alternative domain over which the authors of the DALY could have sought equality would have been in the treatment of the gains and losses attributable to the health state.5 By taking account of the effect of context on the severity of a heath condition, one could achieve an equally egalitarian measure, but over a different domain. Taking context into account in the estimation of the severity weights may reduce the egalitarian flavour of the DALY with respect to resource allocation. On the other hand, it would improve the egalitarian flavour of the DALY with respect to the accounting for the impact of health conditions.

CONCLUSION In the development of the DALY one of the aims was to decouple epidemiology from advocacy. The “decoupling” was based on the presumption that advocacy occurred when individuals or groups overestimated the number of people suffering from a health condition. The correct measurement of the burden of disease, it was argued, would remove the opportunities for advocacy, and decision making could then be based on an objective measure. The enthusiasm with which the DALY has been marketed as one of the measures of population health in the face of robust criticism has given rise to the impression that the process behind the development and the use of the DALY has not always been patent [69]. It may, thus, seem that another form of advocacy has occurred in the development of the DALY that is no less driven by ungrounded passion for the area than the advocacy of interest groups (see note 3 Chapter 1). The data presented in our study contradicts a number of assumptions underpinning the DALY in its current form. The findings are stark, but frankly unsurprising, and the mathematical criticisms of the use of a global severity weight for disaggregated units (such as country, age, and sex) are not concealed by profound and arcane knowledge, but are largely obvious. This reinforces the view that advocacy from a particular perspective drives some features of the DALY’s development. This notwithstanding, advocacy is an important part of any health debate, whether it is a debate around the measurement of population health or around the allocation of resources. Strategies in population health that seek to deny the place of advocacy (a) are implicitly based on one form of advocacy and merely seek to displace other forms, and (b) run the risk of becoming the shibboleth of ‘content-free managers’,

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always able to justify their position because they did what the policy said, not because it was just, fair, or rational. Contrary to the current orthodoxy, and as a purely technical matter, the use of a global severity weight in the DALY (or DALE) prevents the meaningful disaggregation of global burden of disease estimates by region, country, age, sex, etc. Where disaggregated data have been published, (e.g., World Health Report 2000), the authors are, contrary to intention, “comparing apples with oranges”. Once the technical arguments for the deficiencies in the DALY are accepted, it also becomes clear that the social preference arguments for the dismissal of contextual factors are flawed. The arguments conflate social factors as statements of social value (e.g., a rich life is worth more than a poor life”) with contextual factors as a statement about the impact of a health condition (e.g., underdeveloped countries have less general infrastructure to ameliorate the negative impact of health conditions than do developed ones). The social, cultural and environmental contexts in which a health condition occur affect the impact of that condition. The correct estimation of the severity weight for a health condition requires that this contextual information is included6. Unfortunately, the inclusion of contextual information in the estimation of the severity weights will add a level of complexity to the task of calculating the global burden of disease. At the limit, every non-fatal health outcome is unique, “… characterized by the initial pathological process, by its antecedent biological, social, economic causes, by the characteristics of the individual affected and the characteristics of the social milieu and by society’s response to the individual with the condition” [2] (p.22-23).

Nonetheless, there has to be a balance between the assumption that the entire world is one context, and the treatment of each individual as belonging to their own unique context. In effect one wants to find those elements of the context that effect populations not individuals. There is no definitive answer to the question of what defines a single context. Different countries will be more or less homogenous on a range of different social, cultural, and environmental factors. There is almost no research that looks at this aspect of health outcomes. Ignoring the problem, however, is not the answer.

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NOTES 1

For a discussion of the various AEDs, readers are referred to a review by Aiken and Brown[54]. Another article recently appeared that compared an older and newer generation AEDs on quality of life in a developed country population [63]. Two findings that are germane to the discussions here were that the use of the newer lamotrigine resulted in a significantly better quality of life than the use of the older carbamazepine. The second point was that lamotrigine users were significantly more likely to remain in the study than carbamazepine users. Carbamazepine users experienced more cognitive side-effects in general and more general changes in energy levels and affect during the first 4 weeks of treatment. 2 Separate papers on the illness narratives, health status and subjective quality of life are being written up for peer reviewed publications specific to the two conditions. The emphasis in this report is the application of the broad findings to the illustration of context on the measurement of burden of disease rather than on epilepsy or paraplegia per se. 3 Musgrove, who was involved in the writing of the WDR, observed recently that it was indeed “disability” that was used [42]. 4 It should be noted that Erik Nord did suggested the use of “within the skin” when evaluating health states, but this was specifically in the context of OECD nations [65]. Whether Nord believed that ignoring the social context was sensible when evaluating health in all countries is not at all certain. 5 Sen argued that the equality sought by utilitarians, was in “…the equal treatment of human beings in the space of gains and losses of utilities “[68] (p.13). Here the argument is recast in terms of equality in the “counting” of the severity of a health state. 6 Anand and Hanson [27] referred to “compensated” and “uncompensated” weights when writing of severity weights modified by social, cultural, and environmental factors. That is, compensated weights were ones that took context into account and the uncompensated weights were the global severity weights. Murray and Acharya referred to “compensated weights” as “contextualized weights” [25].

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CHAPTER 5 A RESEARCH AGENDA When the World Bank embarked on the World Development Report 1993 project [1] it showed real leadership in the development of a measure of population health. It is unfortunate that the measure (the DALY) has become something of a de facto gold standard and has already begun to drive research and policy agenda, before adequate resources could be directed into testing some of the assumptions on which it was based.1 In spite of the move towards a more multidisciplinary approach in public health, current ways of viewing health and disease at the national and international policy level are based largely on a biomedical model. This devalues the importance of social, cultural and environmental contexts (a) as major factors contributing to the burden of disease, and (b) as points of intervention in improving population health. There is a paucity of data on the impact of health conditions in different social, cultural, and environmental settings. The research described in this report was a small step in that direction. The results, however, highlight the need for further research to improve the usefulness of burden of disease studies, particularly if the current model of measurement using the DALY is to remain the dominant approach. We propose a series of “questions to answer” that map-out a research agenda. The agenda draws on a previous proposal by AbouZahr and Vaughan [30]. However, where they focused specifically on conditions related to reproductive health, we have shown that the issue is much broader, encompassing a diverse range of health conditions. •

What defines a context?

This question would require both empirical and theoretical work and the aim would be to develop techniques for identifying the extent to which, say, socioeconomic status or gender contributed uniquely or in combination to the severity of a particular health condition, and how these factors effect the “base” severity of a health condition attributable to the biomedical features of the condition. This would provide information on the extent to which the modification of a contextual factor could ameliorate the severity of a health condition. Answering this question would also answer the more specific question about whether there exists a “development gradient” that affects health outcomes. Techniques in anthropology and decision analysis could provide a starting point to address this question. •

What is the best method for obtaining valid descriptions of health states within contexts?

Although the research in this report combined qualitative and quantitative techniques in novel ways to capture the experience of a health condition in different contexts, if this approach is to become useful and practical on a wider scale, particularly in developing countries, simple cost-effective techniques will need to be developed. The techniques would have to include sound epidemiology and systematically collected qualitative data. Modification of rapid appraisal techniques may be a suitable starting point for this.

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How can one render contextualised descriptions of a health condition for severity weight estimation? It is not enough to gather the contextualised data on many individuals’ experience of health conditions. Robust techniques need to be developed for rendering the mass of data into pithy yet informative descriptions of the experience of a health condition within a context that can be used in severity weight estimation exercises. The techniques need to be well enough articulated that any number of people could analyse the same data and return substantially the same health state descriptions2. •

How should one estimate the severity weight contribution from the contextual factors?

Techniques would also need to be developed for estimating the severity weights from the contextualised descriptions of the health states. Work is already being done in related areas, such as community-based evaluations looking at health-related quality of life [70] (p.59). The methodological approaches may be directly adaptable. Similarly, if panel estimation is to be used, the methodology employed in the DALY may be adaptable. Some of this work is addressed in medical anthropology, psychology and the economics literature. However, there is still work to be done in the use of expert panels. Whatever process is used needs to be open and patent, and subject to peer review. This research agenda will require the investigation of more health conditions in more countries. Given how little is actually known about the experience of health conditions in different contexts, this becomes an important and valuable exercise in descriptive epidemiology that could have a real impact on our understanding of health. The conditions would have to range across the spectrum in terms of the apparent functional loss, disability and handicap. The development of the methods to collect the data and the data themselves would answer most of the above questions. They would also provide an opportunity to investigate the degree to which the results could be generalised to other health conditions

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NOTES 1

Given the regular revisions, it has been suggested that the DALY is more of a ‘Brass Standard’ – it shines like gold, but tarnishes quickly and needs constant polishing. 2 It is in part because of this need that it is intended to make all the data from this research available on the internet.

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