Psychosocial adjustment of pediatric patients after allogeneic ... - Nature

Bone Marrow Transplantation, (1999) 24, 75–80  1999 Stockton Press All rights reserved 0268–3369/99 $12.00 http://www.stockton-press.co.uk/bmt

Psychosocial adjustment of pediatric patients after allogeneic stem cell transplantation R Felder-Puig, C Peters, S Matthes-Martin, M Lamche, C Felsberger, H Gadner and R Topf St Anna Children’s Hospital, Vienna, Austria

Summary: The purpose of this study was to assess the psychosocial adjustment of patients who had been treated with allogeneic stem cell transplantation (SCT) in our clinic. Selection criteria for patients were to be aged 14–30 years at the time of the follow-up, to be at least 2 years post-SCT and to have a very good knowledge of German. Among 31 eligible patients, 26 participated (84% response rate). The patients were between 15 and 27 years old and were on average 7 years (range 2–13) post-SCT. Research instruments consisted of a demographic questionnaire and various subscales of established psychological measures for which data from a sample of bone cancer survivors and population norms were available. About 35% of patients showed high levels of anxiety, 62% appeared to be extremely sensitive and vulnerable, and 35% showed strong, unfulfilled needs in their love lives. In the other domains tested (self-esteem, family and peer relationships, school/ vocational performance, etc), no noticeable differences were found between the subjects and comparable populations. There was no significant association between psychosocial outcome and demographic features or clinical data. Our results suggest that patients who underwent SCT in their childhood or adolescence are at risk of developing long-term emotional or social problems. Due to the retrospective design of our study and the small sample size, no predictive factors for psychosocial distress could be identified. Keywords: BMT; stem cell transplantation; psychosocial adjustment; quality of life; children

With the rapid development and progress in the disciplines of allogeneic BMT and blood stem cell transplantation (SCT), an increasing number of pediatric transplant recipients evidence long-term disease-free survival. Nevertheless, these medical procedures are still associated with many adverse side-effects, prolonged hospitalization and isolation, risk of persistent treatment-related sequelae and a relatively high possibility of death. These circumstances constitute enormous challenges and place considerable demands on the coping skills, psychological integrity and Correspondence: Dr R Topf, St Anna Children’s Hospital, Kinderspitalgasse 6, A-1090 Vienna, Austria Received 9 October 1998; accepted 17 February 1999

social supports of the children and their parents. Being confronted with a consistent sequence of psychological and physical stressors, there are hardly any patients who do not show in at least one stage of the treatment conspicious behavioral and psychological responses to this strain, including anxiety, depressive mood, withdrawal, hostility, non-compliance, and others.1 In our clinic, an experienced multidisciplinary team tries to supply direct emotional support and provide appropriate interventions in order to reduce the various types of distress.2 However, there always remains some uncertainty if the negative symptoms dissipate after treatment or if they develop into persistent personality disturbances. Reviewing the recent literature, we found a number of reports investigating long-term quality of life and psychosocial adjustment in adult BMT recipients.3–8 Generally, these studies show that post-BMT outcome varies considerably. While some patients evidence good adaptation and relatively normal quality of life, others demonstrate a variety of complications and residual problems. In pediatric BMT or SCT recipients, late physical side-effects of the treatment are well documented,9–11 but issues relating to psychosocial adaptation are relatively unexplored.1 Nespoli et al12 studied 36 children and adolescents who were 1–8 years post-BMT. Life satisfaction, as expressed by patients and their parents, was substantially good and only 16% complained about physical problems. It seemed, however, evident that the BMT experience was accompanied by persistent psychological symptoms. Anxiety and conduct disorders were prevalent in younger subjects whereas older survivors exhibited feelings of depression and inadequacy. The authors also found that younger children demonstrated an overdeveloped sense of perfectionism, which was interpreted as an attempt to control reality and suppress fears of a relapse. Schmidt et al4 assessed the quality of life of 162 adult and 50 pediatric BMT survivors. All respondents were at least 1 year post-BMT. Negative physical symptoms were significantly more prevalent in the adult transplantation population than in the pediatric transplant recipients, including chronic GVHD, frequent colds and skin changes, and the latter group also needed prescribed medication less regularly. Almost all pediatric recipients were attending school or were working at the time of the interview. The subjects were asked to rate their overall quality of life on a scale from 1 to 10 with 10 representing the highest quality. The median score for pediatric patients was 9.5 whereas adult patients showed a median score of 9.0. In a recent study, Badell et al13 evaluated the quality of

Psychosocial adjustment after SCT R Felder-Puig et al

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life of 98 young survivors who were between 3 and 16 years post-BMT. By means of telephone interviews, the authors enquired about problems relating to interpersonal relationships, physical aspects, emotional well-being, occupational situation and health-related concerns. Overall, BMT survivors reported less problems than a control group of 58 healthy subjects of similar age. Major concerns of the BMT sample included physical appearance, academic performance and vocational possibilities. This scarce information about survivors’ quality of life and our interest in the long-term adaptation of patients treated in our clinic led us to assess the psychosocial situation of SCT survivors. Our goal was to provide some more insight into the most common problems that confront these patients after re-entering normal life outside the hospital. Patients and methods We decided to assess the psychosocial adjustment of pediatric SCT recipients with the use of standardized psychological instruments, for which norms were available and which were previously implemented by us in a study on bone cancer survivors.14 This proceeding would permit analysis of the collected data on the basis of the reference values of a comparable clinical group and the general population. The measures which were based on self-rating were only applicable for persons who were at least 14 years old and who had re-entered ‘normal’ life outside the hospital. Thus, to be eligible for participation in the study, the patients had to be between 14 and 30 years at the time of the assessment, to be at least 2 years post-SCT and to be able to read and write German. Subjects who met these criteria were contacted by telephone and invited to come to the clinic to complete a questionnaire after presenting at a medical check-up. The research instruments consisted of various subscales of psychometric measures, which had been developed and tested with large samples (n ⬎ 1000) from German-speaking countries. All of them met the usually required standards for reliability and validity. The scales used in the present study were the following: (1) The scale ‘depressive mood’ from the Questionnaire on Subjective Well-Being.15 Sample items were: ‘I do not feel like doing anything’ or ‘I don’t enjoy doing anything anymore’. Items were rated on six-point scales. Population norms consisted of data from 1901 persons aged 14–20 years. (2) The scale ‘trait-anxiety’ from the State-Trait-Anxiety Inventory.16 Sample items were ‘I feel nervous when thinking of my present concerns’ or ‘I’m often worried about unimportant things’. Respondents rated their agreement on four-point scales. Data on 342 women aged 15–29 years and 244 men aged 15–29 years were used as population reference values. (3) The scales ‘self-esteem’, ‘sensitivity/vulnerability’, ‘ability to mix with others’ and ‘general efficiency’ from the Frankfurt Self-Concept Scales.17 Sample items were: ‘Sometimes I believe that I’m totally useless’, ‘I’m too sensitive’, ‘I’m shy and insecure when being with other people’ or ‘I always find it difficult to realize my plans and

most of the time I fail’. Subjects answered items on sixpoint-scales. The population reference groups comprised 568 persons aged 13–20 years. (4) From the Questionnaire on Life Goals and Satisfaction with Life18 we selected three items each that represented life goals in the areas of ‘love life’, ‘family relationships’, ‘peer relationships’ and ‘school or vocational performance’. The subjects had to first rate on five-point scales how important it would be for them to attain a specific goal (eg ‘How important would it be for you to have a loving relationship? . . . to be successful in your job/at school?). Then they were asked to indicate on five-point scales to what extent they had achieved this goal in their present life (eg ‘Do you have a loving relationship?’ ‘Are you successful in your job/at school?’). The balance score, that was ‘importance’ minus ‘attainment’, reflected the quality of the area. Balance scores ⭓2 indicated severe dissatisfaction with or maladjustment in the respective domain. The authors of the instrument had developed and validated this formula after testing pupils and teachers from seven different German schools (n ⬎ 1000). Raw scores of the scales ‘anxiety’, ‘depressive mood’, ‘self-esteem’, ‘sensitivity/vulnerability’, ‘ability to mix with others’ and ‘general efficiency’ were compared against standardized tables and then converted to percentile scores in order to facilitate comparison across variables. Disturbances in these domains are prevalent in the norm populations of the tests with a rate of 25–30%, so we determined the 30th percentile to be the ‘cutting score’ for differentiation between normally/well-adjusted and maladjusted subjects. For the subscales ‘family relationships’, ‘peer group relationships’, ‘love life’ and ‘school or vocational performance’ of the Questionnaire on Life Goals and Satisfaction with Life, a summary score of ⭓5 for the 2 ⫻ 3 item balance scores (‘importance’ minus ‘attainment’) was defined to be indicative of serious problems in these domains. We analyzed the data for significant differences in the mean scores of SCT and bone cancer survivors. As we had found a high number of subjects scoring along the positive ends of the scales in our study on bone cancer survivors14 and mean scores might obscure important characteristics of the samples, we also determined the incidence of maladjusted persons in both groups using the procedure described above. A self-designed questionnaire enquired about the patients’ marital status, living arrangements, education, occupation, hobbies and specific problems related to disease and therapy. Patients were also asked to grade their overall quality of life on a scale of 1 (= very bad) to 6 (= very good). Clinical data were extracted from the patients’ medical records. Cronbach alpha coefficients were calculated to determine internal consistency of the applied scales. Differences between SCT and bone cancer survivors were evaluated with the use of Student’s t-tests. We computed regression analyses and one-way analyses of variance to test for association between demographic and disease-related variables and each of the psychosocial variables assessed. A P level less than or equal to 0.05 was used as the criterion for statistical significance.


Results All patients meeting the eligibility criteria were approached (n = 31). The final sample consisted of 26 subjects, which reflected a participation rate of 84%. Reasons for not participating were, being unable to locate (n = 3), refusal to participate (n = 1) and mental retardation (n = 1). Sociodemographic and disease characteristics of the subject and control group are summarized in Table 1. Control subjects were on the average 4 years older than SCT survivors, both at diagnosis and follow-up. The average time elapsed since diagnosis was 8 years in both groups. Descriptive statistics were calculated for all psychosocial variables assessed. The subscales showed good internal consistency for the subject group (Cronbach-alphas ⭓0.69), just as they did for the control group.14 SCT survivors demonstrated significantly higher levels of anxiety and sensitivity/vulnerability as well as significantly more problems with regard to love life and school or vocational performance than bone cancer survivors (see Table 2). Whilst the number of maladjusted subjects in the control group did

Table 1 Sociodemographic and disease characteristics of SCT survivors and bone cancer survivors (control group)

No. Gender female male Age mean (years) s.d. range Diagnosis ALL AML SAA CML NHL PNET Ewing Osteo Age of diagnosis mean (years) s.d. range Age of SCT mean (years) s.d. range Time since diagnosis mean (years) s.d. range Time since SCT mean (years) ⬍5 years 5–10 years ⬎10 years Chronic GVHD

SCT survivors (%)

Control group (%)

26

60

14 (53.8) 12 (46.2)

26 (43.3) 34 (56.7)

19.5 3.6 15–27

23.5 4.3 15–30

14 5 3 1 1 1 1

(53.8) (19.2) (11.5) (3.8) (3.8) (3.8) (3.8) 11.0 4.1 3–17

15 (25.0) 45 (75.0) 15.3 5.0 0–25

12.6 3.1 5–18 8.5 4.2 3–19 8 13 5 6

8.2 4.7 2–21

7.0 (30.8) (50.0) (19.2) (23.1)

SCT = allogeneic stem cell transplantation; NHL = non-Hodgkin-lymphoma; PNET = primitive neuro-endocrine tumor; Ewing’s = Ewing’s tumor; osteo = osteosarcoma.

not exceed 30% in any of the variables assessed, the SCT sample demonstrated higher rates of maladjustment in the variables anxiety, sensitivity/vulnerability and love life. Regression analyses and one-way analyses of variance were performed to determine whether the variables gender, age, age of SCT, time since SCT and presence of chronic GVHD were associated with psychosocial outcome. None of these variables demonstrated a significant relationship with the psychosocial variables assessed (P ⬎ 0.05). Analysis of the demographic questionnaires revealed that 25 SCT survivors were single and one was married and that 22 lived with their parents, three lived with a partner and one stayed with his brother. More than half of the subjects (n = 14) were attending school or college. Of the 12 patients who had finished school, four were working, four were apprentices and four were job-less. Questioned about their spare time activities, the patients reported the following most common hobbies: watching TV (31%), reading (27%), listening to music (27%), bicycling (27%) and going out (19%). Most frequent persistent disease- or treatment-related problems were restrictions in sports activities (35%, n = 9) and mobility (19%, n = 5). Catching up with school had been very hard for 38% of patients (n = 10) after discharge from hospital, but except for two all were able to overcome these difficulties. Three patients (12%) said that their problems relating to love life were a consequence of their disease and treatment, and for two patients (8%) therapy had had an adverse impact on their vocational situation. Problems relating to peer relationships, sleeping difficulties and physical appearance were reported by one patient each. Discussion The major questions addressed in this study were whether treatment with SCT during childhood or adolescence has a long-term negative impact on the psychosocial functioning of patients, and which most common problems survivors had to face after reintegration into normal life. Research instruments included a self-designed questionnaire and selected subscales of established psychometric measures of German origin, which allowed for data comparison with population norms and a sample of similarly aged bone cancer survivors, respectively. About 35% of SCT survivors showed high levels of anxiety, 62% appeared to be extremely sensitive and vulnerable, and 35% demonstrated strong, unfulfilled needs in the area of their love life. The level of psychological distress in these domains was significantly higher in the subjects than in bone cancer survivors. SCT survivors were also significantly more dissatisfied with their school or vocational performance than the control group, but the proportion of major problems in this area (12%) was not higher than seen in similarly aged members of the general population. In the other domains tested (depressive mood, self-esteem, ability to mix with others, family and peer relationships, general efficiency), SCT survivors did not score significantly different from the control group, nor was the rate of maladjusted subjects higher than in the general population. Overall quality of life was rated, on average, as ‘good’ (mean ⬇ 5 on a scale of 1 to 6) by both SCT and bone cancer survivors.

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Table 2

Psychosocial adjustment of SCT survivors and bone cancer survivors (control group)

Dimension of psychosocial adjustment

SCT survivors Cronbach-␣

Anxiety Depressive mood Self-esteem Sensitivity/vulnerability Ability to mix with others Family relationships Peer group relationships Love life General efficiency School/vocational performance Overall quality of life

0.90 0.84 0.87 0.71 0.75 0.71 0.73 0.73 0.86 0.69

Control group

Mean (s.d.)

Mean (s.d.)

46.2a 58.1a 63.4a 23.8a 60.0a 1.7b 2.3b 4.2b 64.3a 2.7b 5.1a

60.2a 55.0a 76.3a 62.0a 67.1a 1.2b 1.5b 2.1b 72.8a 1.7b 5.3a

(30.3) (30.4) (33.0) (19.9) (32.6) (1.5) (2.6) (2.3) (33.4) (2.2) (1.0)

(30.0) (25.7) (28.6) (33.6) (30.8) (1.5) (1.9) (3.0) (29.0) (1.7) (0.8)

Rates of maladjustment (%)

P value

SCT-/control group

0.05* 0.63 0.07 ⬍0.001** 0.34 0.17 0.11 0.001** 0.24 0.03* 0.67

35/22 23/21 23/12 62/26 15/17 0/4 15/5 35/10 23/14 12/9

a

Lower scores denote greater psychosocial dysfunction. Higher scores denote greater psychosocial dysfunction. *Significant at the P ⭐ 0.05 level. **Significant at the P ⭐ 0.01 level.

b

The comparability of our findings to results from previous studies on pediatric BMT survivors is complicated by the fact that all used different methodology and evaluation methods. The adolescent patients in the study of Nespoli et al12 showed slightly increased levels of depression and feelings of inadequacy as compared to Italian population norms. Further, female teenagers in the BMT sample had significantly more difficulties in coping with themselves and significant others than a control group of healthy girls. Schmidt et al4 focused on the assessment of physical sequelae and did not implement any psychological tests but confined themselves to asking one single question about overall quality of life. The majority of their 50 pediatric BMT subjects showed positive perceptions of their quality of life, equally to our SCT subjects. Badell et al13 used a short, self-designed questionnaire for the evaluation of quality of life in pediatric BMT survivors. Their 98 subjects gave significantly higher quality of life ratings and reported fewer problems in various aspects of life than a control group of healthy peers. All these findings lead to the conclusion that the majority of BMT or SCT survivors demonstrate, in general, good quality of life, but may also exhibit some kind of psychological distress which exceeds the normal. The counter-intuitive finding of Badell et al that survivors do even better than healthy peers was also reported in some studies on childhood cancer patients.19–21 When interpreting these results, one has to consider the presence of some uniquely adaptive coping behaviors in these patients. In fact, there are a number of studies suggesting that pediatric oncology patients mobilize denial in response to questioning about their life situation and emotional state, and are often not aware of or refuse to acknowledge problems.22–25 We therefore believe we have used an appropriate primary control group, especially since pediatric bone cancer patients also receive a particularly burdening treatment including multi-modality therapy and are faced with a risk of permanent physical handicap or disfigurement. Our study revealed markedly high levels of anxiety, sensitivity/vulnerability and dissatisfaction with their love life in SCT survivors. Anxiety is probably the most fre-

quently observed psychological response to the multiple stressors during SCT treatment. Repeated anxiety-inducing procedures and situations and traumatic experiences during therapy as well as fears of relapse, which remain prevalent, often on a subconscious level, even years after therapy, may lead to the development of ‘trait-anxiety’. The same factors possibly cause increased levels of sensitivity and vulnerability persisting over time. An individual with a low score in this variable is described in the test manual as someone being rarely cheerful, very sensitive and easily hurt. The author of the test recommends further diagnostic check-up and, if necessary, consequent psychotherapy in order to increase the individual’s psychological stability.17 Little is known about the love life of pediatric BMT or SCT survivors. Decreased sexual functioning has been described in adult transplant recipients including diminution of sexual interest, enjoyment, satisfaction or activity, and some physical dysfunction.26,27 Inspection of the individual responses in the scale ‘love life’ revealed that many of our subjects missed having a love relationship. Increased anxiety of getting involved with someone, less opportunities in meeting a potential partner or having an inferior body image as compared to healthy peers are possible explanations. Research on neuropsychological functioning of pediatric BMT recipients and/or childhood cancer patients showed long-term cognitive deficits, with CNS irradiation or TBI and age at treatment being influencing factors.28,29 Almost all subjects in our sample reported satisfactory academic performance, although determined efforts were necessary to catch up with school after treatment. This may be taken as evidence of survivors’ ability to concentrate on schooling in spite of possible handicaps. Overall, 22/26 patients (85%) had been able to return to school or employment. This concurs roughly with the data of Schmidt et al4 who reported that 95% (47/50) of their pediatric BMT survivors were attending school or employed at follow-up. Of our four job-less subjects, two attributed unemployment to disease or treatment sequelae and the other two gave no reason for their vocational situ-


ation. However, as our sample included only 12 patients who had finished school, the number of subjects facing job difficulties or being unable to work is too small to make any reliable inferences. This issue certainly needs further investigation. Restrictions in sports activities and mobility were the main problems reported and attributed to disease or treatment sequelae. SCT survivors were more likely to perform spare time activities which required no physical activity, such as watching TV, reading or listening to music. For comparison, bone cancer survivors had reported a remarkable amount of sports activity including bicycling (37%), swimming (35%), tennis (12%) or skiing (10%).14 Sociodemographic and disease-related variables, such as age of SCT, time since SCT and presence of chronic GVHD, were not predictive of psychosocial distress. We did not control for other important disease and treatment variables, as our sample was not large enough to simultaneously consider the impact and interaction of the possible factors influencing survivors’ adjustment. There is some evidence in the research on childhood cancer patients that clinical data contribute less to the psychosocial development of survivors than do factors such as coping styles or communication patterns.30 Koocher and O’Malley31 concluded in their pioneering work that ‘survivors who adjust well do so regardless of the degree of residual physical impairment’. Although the participation rate of 84% reflects a high likelihood of having obtained a reasonably unbiased sample, the generalizability of our findings is limited by the small sample size and the heterogeneity of the sample with regard to age and interval between transplantation and follow-up. Data were based on self-assessment and may therefore be subject to some social desirability bias,32 ie in reality, there are possibly more problems, which have been, deliberately or unconsciously, not reported by our subjects. Our findings add to the scarce up-to-date information available on the long-term psychosocial adjustment of pediatric patients receiving allogeneic BMT or SCT. Despite minor methodological limitations, they indicate that these patients are at risk of developing psychosocial sequelae. Prospective assessments are needed to find out at what stage of treatment which types of psychological distress manifest and under which circumstances these symptoms dissipate or result in persistent disturbances. Specific areas that require additional research include survivors’ ability to achieve academic and career goals, to establish a satisfying loving relationship with a partner and to perform ageappropriate activities and lifestyles. Another important issue would be to identify variables such as family support, coping styles or communication patterns during treatment that are associated with post-SCT psychosocial adjustment and quality of life.1

3

4 5

6

7 8

9

10 11 12 13

14 15 16 17 18 19

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