Arthritis Care & Research Vol. 63, No. 6, June 2011, pp 899 –908 DOI 10.1002/acr.20462 © 2011, American College of Rheumatology
Domains of Health-Related Quality of Life Important and Relevant to Multiethnic EnglishSpeaking Asian Systemic Lupus Erythematosus Patients: A Focus Group Study YEN LING MANDY OW,1 JULIAN THUMBOO,2 DAVID CELLA,3 YIN BUN CHEUNG,4 KOK YONG FONG,2 AND HWEE LIN WEE1
Objective. To identify health-related quality of life (HRQOL) domains of importance to multiethnic Asian systemic lupus erythematosus (SLE) patients, to identify content gaps in existing SLE-speciﬁc HRQOL measures, and to determine whether the Patient-Reported Outcomes Measurement Information System (PROMIS) item banks could serve as a core set of questions for HRQOL assessment among SLE patients. Methods. English-speaking patients with physician-diagnosed SLE from a specialist clinic in a tertiary care hospital in Singapore and a patient support group were recruited. Thematic analysis was performed to distill themes from transcripts through open coding by 2 independent coders and axial coding for reﬁnement of categories. Items from 3 existing SLE-speciﬁc measures and PROMIS Version 1.0 Item Banks were compared with identiﬁed subthemes. Results. Twenty-seven female and 2 male participants (21 Chinese, 4 Malay, 3 Indian, 1 other) ages 23– 62 years participated in 6 focus groups and 2 individual interviews, respectively. Twenty-one domains and 92 subthemes were identiﬁed. Domains of family, relationships, stigma and discrimination, and freedom were unaddressed by existing SLE-speciﬁc measures. Forty subthemes from 14 domains were addressed by the PROMIS Version 1.0 Item Banks (Physical Function, Pain, Fatigue, Sleep Disturbance, Sleep-Related Impairment, Anger, Anxiety, and Depression banks). Conclusion. Family and stigma and discrimination (identiﬁed as content gaps) may be accentuated in the Asian sociocultural context. PROMIS item banks have tremendous potential to serve as a core set of items for HRQOL assessment in SLE patients. Additional items may be written to ﬁll the gaps in existing PROMIS item banks.
INTRODUCTION Systemic lupus erythematosus (SLE) is an autoimmune disease with variable clinical manifestations and an unSupported by the National Medical Research Council (Singapore) New Investigator Grant (NMRC/NIG/0039/2008). 1 Yen Ling Mandy Ow, BSc, Hwee Lin Wee, PhD: National University of Singapore, Singapore; 2Julian Thumboo, MBBS, MRCP, MMed, FAMS, FRCP, Kok Yong Fong, MBBS, MMed, FAMS, FRCP: Singapore General Hospital and National University of Singapore, Singapore; 3David Cella, PhD: Northwestern University Feinberg School of Medicine, Chicago, Illinois; 4Yin Bun Cheung, PhD, CStat: Singapore Clinical Research Institute and Duke-NUS Graduate Medical School, Singapore. Dr. Cella has received consultant fees, speaking fees, and/or honoraria (less than $10,000 each) from Genentech, Bayer, Novartis, and GlaxoSmithKline, and (more than $10,000) from Pﬁzer. Address correspondence to Hwee Lin Wee, PhD, National University of Singapore, Block S7, #02-05, 18 Science Drive 4, Singapore 117543. E-mail: [email protected]
Submitted for publication October 17, 2010; accepted in revised form February 28, 2011.
predictable, ﬂuctuating disease course (1). Health-related quality of life (HRQOL) among SLE patients is reduced (2) as SLE has an impact on the physical, social, and psychological aspects of life (2–5). Therefore, a challenge in outcome assessment among patients with SLE is in identifying domains that are relevant and important because of the heterogeneous manifestations of SLE (6). Also, in understanding the impact of SLE, the importance of patient perspectives in various cultures has been recognized (5,7). Input from SLE patients in Western countries has been sought in recent qualitative studies (3–5,8 –12). These include studies for the development of SLE-speciﬁc HRQOL instruments, the L-QoL and the LupusQoL (8,9), and for the development of the International Classiﬁcation of Function, Disability and Health core sets for SLE (3,5). There is one exception: the Systemic Lupus Erythematosus–speciﬁc Quality of Life (SLEQOL), which was developed in Singapore (13). However, contents of the SLEQOL were derived from rheumatologists and nurse clinicians without a formal qualitative study involving patient input. The developers of the SLEQOL have acknowledged the lack of a formal qualitative study as an important limitation of the study. 899
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Signiﬁcance & Innovations ● Four domains have been identiﬁed as content gaps in existing systemic lupus erythematosus (SLE)–speciﬁc health-related quality of life (HRQOL) measures, speciﬁcally family, relationships, freedom, and stigma and discrimination. Two of these domains, family and stigma and discrimination, may be accentuated in the Asian sociocultural context. ● Forty subthemes from 14 domains identiﬁed in this study as important and relevant to SLE patients were addressed by the PatientReported Outcomes Measurement Information System (PROMIS) Version 1.0 Item Banks. PROMIS item banks have tremendous potential to serve as a core set of items for HRQOL assessment in SLE patients. ● Disease-speciﬁc item banks would provide additional value to PROMIS banks in the assessment of domains that are signiﬁcant across many but not all diseases, such as stigma.
SLE-speciﬁc HRQOL measures address additional domains relevant to SLE patients that are not addressed in generic measures, such as the Medical Outcomes Study Short Form 36 (2,14). These domains include fatigue, sleep disturbances, sexual functioning, and body image (14). Four SLE-speciﬁc patient-reported outcome measures are available for use in adult SLE patients: the SLEQOL (13), the LupusQoL (9), the L-QoL (8), and the SLE Symptom Checklist (SSC) (15), which contains a list of symptoms and is not considered to be a true HRQOL measure (14). Although well validated, both generic and SLE-speciﬁc measures experience the limitations of “one size ﬁts all” instruments because the same items are administered to all patients (16). A newer approach involving item-response theory (IRT) has demonstrated advantages for clinical outcome assessment (17). IRT is a family of measurement models that places each item measuring a latent trait on a common metric, according to the probability of a positive response (18). An important application of IRT is the use of computerized adaptive testing, whereby selective administration of items tailored to the individual’s ability level contributes to increased precision yet lower respondent burden (19). Items are also selected from pools covering a wider range of difﬁculty (17). Patient-Reported Outcomes Measurement Information System (PROMIS), a National Institutes of Health initiative, has started construction of item banks to measure key symptoms and health concepts applicable to a range of chronic conditions (20). This effort was undertaken to standardize and promote a common measurement system for patient-reported outcomes across clinical research (20). Five domains were selected as initial areas for PROMIS item bank construction: 1) physical functioning, 2) fatigue, 3) pain, 4) emotional distress, and 5) social role participation (20). Items from these 5 domains are available online in the form of PROMIS Version 1.0 Item Banks (21). Because of culture-speciﬁc differences in HRQOL (22), it is unknown whether domains identiﬁed by Western patients are also important to Asian patients. Additional
domains important to Asian patients may also be elucidated. Therefore, Asian patient perspectives are required for development of an instrument relevant for use in Asian SLE patients. Our ﬁrst two objectives were to identify HRQOL domains of importance to SLE patients in Singapore and to identify content gaps in existing SLE-speciﬁc HRQOL measures. As PROMIS item banks are meant to be relevant across chronic diseases, existing banks could potentially be used for HRQOL assessment among SLE patients. To avoid replication in the construction of item banks, our third objective was to determine whether PROMIS item banks could serve as a core set of questions for the assessment of HRQOL among SLE patients.
PATIENTS AND METHODS Participants. In this institutional review board–approved study, patients with SLE were recruited from an outpatient clinic at the Singapore General Hospital and from a patient support group, the Lupus Association (Singapore). Inclusion criteria for the study were 1) a diagnosis of SLE made by a rheumatologist, 2) age 21– 65 years, 3) able to read and understand English, 4) able to speak coherently and provide written informed consent, and 5) able to travel to the interview venue without assistance. Due to the relatively low prevalence of SLE, a small patient number contributed to an a priori decision to stratify participants only by sex, as it was anticipated that sensitive issues may be discussed. However, participants were selected for each session based on their availability and characteristics, such as age and ethnicity, to be as homogenous as possible. Recruitment was ongoing and focus group sessions were convened whenever sufﬁcient numbers were available for a session. Procedures. We aimed to recruit 4 to 7 participants for each session. The ideal focus group composition is 4 to 12 participants, with the optimal number dependent on the aim of the research study (23). Interaction between focus group members and the moderator can generate in-depth and more diverse information than other methods, such as surveys or individual interviews (24). More importantly, the discussion may proceed with relatively little input from the moderator (25), generating more patient-related content. Based on the phenomenological approach to study the experience of SLE patients through the eyes of the participants (26), the interview guide was designed to be open ended. It proceeded from the most general to speciﬁc questions, minimizing the inﬂuence of moderator probes. A 60-minute discussion on SLE-related impact on HRQOL was led by a moderator and facilitated by a note taker. The ﬁrst question for discussion was, “In general, how has having SLE affected your health-related quality of life?” Subsequent questions focused the discussion on the 3 broad domains of physical, mental, and social well-being according to the World Health Organization deﬁnition of health as a state of physical, mental, and social well-being (27). Because we were unable to recruit sufﬁcient male participants to form a focus group (due largely to the female preponderance in SLE, and partly to the varied work
HRQOL Domains Important to Multiethnic Asian SLE Patients
Table 1. Participant characteristics (n ⴝ 29)* Value Age, median (range) years 45 (23–62) Sex† Female 27 (93) Male 2 (7) Race Chinese 21 (72) Malay 4 (14) Indian 3 (10) Other 1 (3) Marital status Single 8 (28) Married 20 (69) Divorced 1 (3) Work status Working 17 (59) Homemaker 7 (24) Retired 1 (3) Unemployed (able to work) 1 (3) Unemployed (unable to work) 1 (3) Other 2 (7) Education University degree 10 (34) Diploma 4 (14) GCE “A” levels 5 (17) GCE “N” or “O” levels 6 (21) Incomplete secondary schooling and below 4 (14) Years since diagnosis of SLE ⬍5 8 (28) 5–10 2 (7) 11–20 10 (34) ⬎20 9 (31) * Values are the number (percentage) unless otherwise indicated. GCE ⫽ General Certiﬁcate of Education; A ⫽ Advanced; N ⫽ Normal; O ⫽ Ordinary; SLE ⫽ systemic lupus erythematosus. † Women participated in 6 focus group sessions. Men participated in individual interviews.
schedule of the patients), semistructured individual interviews were conducted by a male interviewer based on the discussion guide. Following the discussion, participants were asked to rate 7 areas of life as “highly,” “somewhat,” or “not at all” important and relevant to them. These areas were preselected based on a review of domains addressed by existing SLE-speciﬁc instruments and the PROMIS item banks. All focus groups, interviews, and subsequent data analysis were conducted in English. Participants also completed a questionnaire on background information and were presented with a token sum to cover the cost of travel and for their participation. Data analysis. Focus group and interview discussions were voice recorded, transcribed verbatim, and analyzed using thematic analysis. The open-ended discussion guide and data-driven analytic methods adopted in this study are based on elements of the grounded theory to encourage development of conceptual frameworks that are derived from participant input, rather than existing concepts (26). In thematic analysis, segments of texts describing similar
901 manifestations are grouped into themes (28). Two independent analysts, trained graduate students who were unfamiliar with existing frameworks of HRQOL, read the transcripts reﬂectively to identify relevant categories and assign codes to text segments in a process known as open coding (29). Open coding is a data-driven process that identiﬁes themes from data directly, rather than using ideas from existing literature or frameworks (30). A codebook comprising 5 ﬁelds (code title, deﬁnition, examples, guidelines for use, and relationship to other codes) was created as analysts coded subsets of transcripts in an unspeciﬁed number of rounds. This iterative process of independent coding and consensus meetings continued until the investigators were satisﬁed that the codes could be consistently applied. The codes were then independently applied to all of the transcripts and coding discrepancies were resolved at a ﬁnal consensus meeting. Further analysis was facilitated by axial coding using Atlas.ti, a qualitative analysis software (31). Axial coding is the process of conﬁrming what has been established and developing it to a more abstract level (32). The ﬁnal output of thematic analysis was a list of themes descriptive of HRQOL domains and their constituent subthemes, which elaborated on the scope of the domains as discussed by study participants. Mapping of themes and subthemes to SLE-speciﬁc instruments and PROMIS Version 1.0 Item Banks. Existing SLE-speciﬁc instruments were identiﬁed through the PROQOLID database and a systematic literature search using PubMed with the terms “systemic lupus erythematosus” AND “quality of life”. Four instruments were identiﬁed from the search and items from the SLEQOL, the LupusQoL, and the SSC were compared with subthemes identiﬁed from the focus groups because the developers of these instruments did not object to us doing so (9,13,15). The SLEQOL and the LupusQoL are SLE-speciﬁc HRQOL measures (9,13). The SSC is a checklist of symptoms covering physical symptoms and one item on mood changes (15). Items from 11 of the 12 PROMIS Version 1.0 Item Banks (excluding global health) were also compared with focus group subthemes. These 11 banks covered the 5 domains selected as the initial areas for PROMIS item bank construction (physical functioning, fatigue, pain, emotional distress, and social role participation) (21). A content gap was identiﬁed when subthemes and domains elicited from focus groups were not addressed by any item from these existing sources.
RESULTS Focus group participants. Twenty-seven female SLE participants ages 23– 62 years participated in 6 focus groups conducted from August 2009 to March 2010. Four sessions comprised 4 to 5 participants and the other sessions comprised 2 participants (due to unanticipated nonattendance at very short notice) and 7 participants each. Saturation of topics occurred at the ﬁfth session, with no new topics emerging at the sixth session. In addition, 2 male SLE participants ages 32 and 39 years participated in individual interviews. The majority of participants were
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Table 2. Domains and subthemes important to SLE patients in Singapore and extent of coverage by existing SLE-speciﬁc instruments and PROMIS Version 1.0 Item Banks* Concepts derived from the focus groups (broad category codes, domains, and subthemes)
Physical symptoms Fatigue I feel tired Fatigue limits the work I can do Fatigue limits the activities I participate in I need to have early nights I feel like I can’t get out of bed I had problems concentrating Others perceive that I am lazy because of my fatigue Pain I feel pain My pain was so bad I would do anything to end it Pain affects my mobility Pain limits the work I can do Pain limits the activities I can participate in Pain makes me feel depressed Pain has set me back in life Sleep disturbance I have difﬁculty falling asleep I cannot get a sound sleep I have interrupted sleep I do not feel the need to sleep at bedtime I sleep at odd hours Poor sleep affects my ability to work in the day I feel frustrated about not being able to sleep Activities ADL I have difﬁculty moving around the house I have difﬁculty feeding myself I have difﬁculty dressing myself I have difﬁculty with basic, everyday tasks I have difﬁculty performing household chores I am dependent on others for my daily activities Leisure activities (non-ADL) I am unable to do things I would like to for leisure I refrain from certain leisure activities I refrain from going to sunny places for leisure I restrict myself in the choice of holiday destinations Social activities I am unable to take part in social activities I want to I refrain from participating in certain social activities I would rather not participate in social activities Social life Family My willingness to start a family has been affected My ability to start or expand my family has been affected My ability to carry out duties to family members has been affected Emotional state Anger I feel angry I nitpick on things I have a bad temper I am frustrated I get irritated
Chinese (72%), the others being Malay (14%) or Indian (10%) (Table 1). The median age was 45 years (interquartile range [IQR] 33–53 years) and the median disease du-
FAT FAT FAT FAT
PBE PIN PIN PIN PIN SLP SLP SLP SLP
⫹ ⫹ ⫹ ⫹
PF PF PF PF PF DSA DSA
ANG ANG ANG ANG (continued)
ration was 14 years (IQR 8 –21 years). Most participants were working (59%) or homemakers (24%). Education levels of participants were well varied among degree holders
HRQOL Domains Important to Multiethnic Asian SLE Patients
Table 2. (Cont’d) Concepts derived from the focus groups (broad category codes, domains, and subthemes) Sad I feel sad I feel guilty I feel helpless I feel hopeless I feel hurt by others I feel like I don’t want to live Lonely I feel lonely I feel that no one understands me Fear I feel fearful Worry I feel worried I feel stressed Other Self-esteem I feel humiliated I have low self-esteem Work I am unable to perform the full range of duties or activities I am unable to do as much work as before I refrain from doing too much work because I am afraid of stressing my body out I have less motivation at my job My job prospects are poor because of my illness Taking time off work/school has affected my work negatively I do not perform as well at work I earn less money because I am unable to do as much work as before My employer has a negative impression of me because of my illness
DEP DEP DEP DEP DEP DEP
DEP SR SR SR
⫹ ⫹ ⫹
* SLE ⫽ systemic lupus erythematosus; PROMIS ⫽ Patient-Reported Outcomes Measurement Information System; SLEQOL ⫽ Systemic Lupus Erythematosus–speciﬁc Quality of Life; SSC ⫽ SLE Symptom Checklist; ⫹ ⫽ subtheme/domain is covered by items from existing SLE-speciﬁc measures (LupusQoL, SLEQOL, SSC); FAT ⫽ fatigue; SLI ⫽ sleep-related impairment; PBE ⫽ pain– behavior; PIN ⫽ pain–interference; SLP ⫽ sleep disturbance; ADL ⫽ activities of daily living; PF ⫽ physical function; DSA ⫽ satisfaction with social participation (discretionary social activities); DEP ⫽ depression; SR ⫽ satisfaction with social participation (social roles); ANG ⫽ anger; ANX ⫽ anxiety.
(34%), postsecondary education (31%), secondary education (21%), and below (14%). Thematic analysis. A total of 40 open codes were created, categorized as codes descriptive of an impact on HRQOL (27 codes) or not descriptive of an impact on HRQOL (7 codes), or broad category codes that subsumed other codes (6 codes). Broad category codes were “physical symptoms,” “physiological changes,” “activities,” “social life,” “emotional state,” and “others.” Codes not descriptive of an impact on HRQOL, such as “coping with SLE,” were created to facilitate understanding of the impact of SLE on HRQOL and their relationships with other themes. Open codes descriptive of an HRQOL impact were further reﬁned into 21 domains comprising 92 subthemes during axial coding. A ﬂow chart and table illustrating the processes of open coding, axial coding, and mapping to existing items are available in Supplementary Appendix A (available in the online version of this article at http:// onlinelibrary.wiley.com/journal/10.1002/(ISSN)2151-4658).
Domains of HRQOL identiﬁed. Domains identiﬁed by participants were classiﬁed into 5 categories: physical symptoms, activities, social life, emotional state, and other. Because PROMIS Version 1.0 Item Banks address only selected domains, results of subtheme mapping to existing items are shown in separate tables (Tables 2 and 3). Table 2 lists only domains addressed by PROMIS banks. Domains addressed by PROMIS Version 1.0 Item Banks. Domains grouped under “physical symptoms” (fatigue, pain, and sleep disturbance) and “emotional state” (anger, sad, lonely, fear, and worry) were generally addressed by the LupusQoL, SLEQOL, and relevant PROMIS banks. Items from the PROMIS banks covered the most number of subthemes within these domains. Domains within “activities” were activities of daily living, leisure activities, and social activities. Activities of daily living are addressed by the SLEQOL, LupusQoL, and items from the PROMIS Physical Function bank. Unaddressed subthemes of leisure and social activities were related to restrictions that
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Table 3. Domains and subthemes important to SLE patients in Singapore and extent of coverage by existing SLE-speciﬁc instruments only (i.e., not covered by PROMIS Version 1.0 Item Banks)* Concepts derived from the focus groups (broad category codes, domains, and subthemes) Physical symptoms Other symptoms Tremors Weakness Pain: speciﬁc locations Decreased interest in sex Changes in appearance Social life Relationships I have less social contact with the outside world I prefer to keep things from others I feel that people don’t understand my condition My relationships are strained Other Burden to others I feel that I am a burden to others I am afraid that others will perceive me as a burden Dependence I am dependent on others for my activities of daily living I am afraid that no one will provide care when dependent I do not wish to be dependent on others Stigma and discrimination People avoid or shun me Family or friends exclude me from their activities People make unkind or insensitive remarks about me People treat me differently because I have a disease Self-image I look unattractive I get bothered by comments about appearance I am frustrated by my appearance changes My appearance makes me avoid social situations I am more image conscious Freedom There are many restrictions on the things I can do There are many restrictions on the places I can go I cannot do things freely Having to be cautious affects my enjoyment of life Lupus holds me back in life
⫹ ⫹ ⫹
⫹ ⫹ ⫹
* See Table 2 for abbreviations.
SLE patients face in their choice of activities and destinations, rather than being unable to perform them. This was reportedly attributable to the need for minimizing disease ﬂare triggers through avoiding the sun and vigorous activities. From the family domain, only the subtheme of “ability to carry out duties to family members” was covered by the PROMIS Satisfaction with Social Participation (Social Roles) bank. “Willingness to start a family” describes the impact of SLE on participants’ decisions regarding marriage and having children, owing to concerns of disease unpredictability and being a burden to the family. Concerns with childbearing difﬁculties and ﬁnding a partner accepting of one’s medical condition were subsumed under the subtheme “ability to start or expand my family.” The domain “self-esteem” is covered by the LupusQoL, SLEQOL, and the PROMIS Depression bank. Many subthemes emerged within the work domain, reﬂecting the
profound impact of SLE on a patient’s work life. The majority of these subthemes did not map to any existing instrument or item bank. Domains not addressed by PROMIS Version 1.0 Item Banks. Subthemes of symptoms not covered by PROMIS banks included tremor, weakness, pain at speciﬁc locations, decreased interest in sex, and changes in appearance. With the exception of tremors, all symptoms were included in at least one existing SLE-speciﬁc measure. “Burden to others” and “self-image” were covered by the LupusQoL and the SLEQOL, and the LupusQoL contained an item on “dependence.” Other domains, speciﬁcally relationships, stigma and discrimination, and freedom, were generally not covered by existing instruments. Sample quotations illustrating each subtheme from these domains are shown in Table 4. Subthemes of “stigma and discrimination” include being avoided or shunned, being ex-
HRQOL Domains Important to Multiethnic Asian SLE Patients
Table 4. Sample quotations from focus group discussions* Sample quotation Subthemes from domains not addressed by existing SLE-speciﬁc instruments and PROMIS Version 1.0 Item Banks Relationships I have less social contact with the outside world I prefer to keep things from others I feel that people don’t understand my condition My relationships are strained
Stigma and discrimination People avoid or shun me
Family or friends exclude me from their activities People make unkind or insensitive remarks about me People treat me differently because I have a disease Freedom There are many restrictions on the things I can do There are many restrictions on the places I can go I cannot do things freely
Having to be cautious affects my enjoyment of life Lupus holds me back in life
Male participants describing and relating the various impact of SLE to work Changes in appearance
I feel fearful
Taking time off work/school has affected my work negatively
“The one year plus I was in hospital—my closest friends, I still keep in touch, but the rest I just cut off. Like now, I don’t really go out and meet them. I don’t like to go out in public” (age 23 years, female, Malay) “I’ll stop seeing some of my friends—I don’t want them to look at my ugly side and I don’t like to explain much” (age 33 years, female, Chinese) “You want to ask for help but you cannot every now and then ask somebody to help you. People think you are so petty, a little bit of pain [and you ask for help] – because they don’t know what is lupus” (age 48 years, female, Malay) “In a way, it has also affected my marriage. That’s the period where men have physical needs and I totally neglected that area of his life because at the point, it was just self-pity, anger, and the grieving process” (age 59 years, female, Chinese) “My face will be puffy and very red. If I wait for a lift, I ﬁnd that my neighbors all avoid me, they don’t want to go in the same lift as me” (age 62 years, female, Chinese) “They think this is contagious. Even my family, my sister-in-law, said ‘you take your food separately’” (age 55 years, female, Chinese) “You will have aunties that go, ‘Wow, why is your face like that?’ I know they mean well but for someone who feels rotten by herself, what kind of stupid comment is that?” (age 31 years, female, Chinese) “My colleagues or friends, who know I have lupus, they will see me in a different light, they see as very fragile. I don’t like any special treatment” (age 39 years, female, Chinese) “[I feel] angry because I’m not able to do things because of the disease. I used to do a lot, you know, there are a lot of restrictions” (age 53 years, female, Chinese) “When I plan for places to go, I will surely think of whether there are medical facilities easily available and sunlight and all these things, there are a lot of restrictions, so have to plan” (age 33 years, female, Chinese) “A lot of things, activities are being put to a stop. You have to pause a while and think, ‘Can I go or not?’ [others] can just go freely” (age 59 years, female, Chinese) “You can’t enjoy as much as before. You think a lot, like, ‘If too long under the sun, will I get affected again?’ It’s like you will be worried and you can’t really enjoy as much as before” (age 30 years, female, Chinese) “Since I’ve got lupus, I’ve got to slow down. Because as you move too hard or too harsh, your body starts to feel the pain. And then you start to have a butterﬂy rash, that’s for me. I have to cool down my activities daily, and my work force” (age 48 years, female, Malay)
“Rash only causes a lot of inconvenience because I still need to face the public at work. Sometimes they will ask, ‘Do you have a skin problem?’” (age 39 years, male, Chinese) “I fear that when the ﬂares and attacks come, these cause a lot of medical fees and money to be spent. I also take a long leave from work, so sometimes I have a fear I will have to give up my job. From there, it affects my family because there’s no income” (age 39 years, male, Chinese) “As long as it’s not a ﬂare that I have to stay in the hospital for quite a long time, like two years back [and] rest for 2 to 3 months at home, that will affect the job. That’s the only stress but if the company is good, they will keep you” (age 32 years, male, Chinese)
* SLE ⫽ systemic lupus erythematosus; PROMIS ⫽ Patient-Reported Outcomes Measurement Information System.
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Table 5. Participant rating of importance and relevance on select areas of life (n ⴝ 29)*
Fatigue Physical function Pain and other symptoms Burden to others Self-image Emotional health Social and occupational activities
No. (%) rated as “highly important”
No. (%) rated as “highly relevant”
18 (62) 17 (59) 16 (55) 13 (45) 10 (34) 12 (41) 7 (24)
20 (69) 10 (34) 14 (48) 10 (34) 10 (34) 9 (31) 6 (21)
* Areas of life were selected for rating based on a review of domains included in existing systemic lupus erythematosus–speciﬁc instruments and Patient-Reported Outcomes Measurement Information System Version 1.0 Item Banks.
cluded from activities, and receiving unkind or insensitive remarks from others. These relate to one’s appearance changes and misconceptions about the disease (such as lupus being contagious or that one has a sexually transmitted disease). Participants also expressed a dislike for being treated differently because of one’s illness. Finally, “freedom” comprised subthemes that described restrictions in activities and places, not being able to do things freely, diminished enjoyment of life, and a slower pace of life. Feelings of restrictions, diminished enjoyment, and a sentiment of being held back in life stemmed from the worry of triggering a disease ﬂare. Domains of HRQOL identiﬁed by male participants. Similar to female participants, male participants (n ⫽ 2) reported symptoms, impact on emotional states, activities, and work. Two main differences were identiﬁed in the accounts of male participants. First, they reported additional challenges of lupus in coping with the physically demanding and outdoor nature of their work. Second, male participants often related an impact of how it affected one’s work, income, and ability to provide for the family, as illustrated with 3 quotes from male participants (Table 4). Rating of importance and relevance on select areas of life. Of the 7 areas of life identiﬁed from existing SLEspeciﬁc instruments or PROMIS Version 1.0 Item Banks, fatigue was rated as “highly important” (62%) and “highly relevant” (69%) by the largest proportion of respondents (Table 5). Two other areas of physical health, speciﬁcally “physical function” and “pain and other symptoms,” were next most frequently rated as highly important and relevant. Comparatively, burden to others and self-image, areas of life addressed by the SLEQOL and the LupusQoL but not PROMIS Version 1.0 Item Banks, were rated “highly relevant” as frequently as physical function (34%).
DISCUSSION This qualitative study identiﬁed domains of HRQOL relevant and important to multiethnic Asian SLE patients in
Singapore using a data-driven analytic methodology. Content gaps from existing SLE-speciﬁc instruments were identiﬁed and the relevance of PROMIS Version 1.0 Item Banks to SLE patients was assessed. Many domains of HRQOL captured by the existing disease-speciﬁc HRQOL instruments were also found to be relevant to the current study’s participants, with the exception of 1) items on speciﬁc symptoms from the SSC and SLEQOL, 2) items on inconvenience of daily medication and clinic visits, 3) items on increased consumption of alcohol or tobacco from the SLEQOL, and 4) items related to the inability to plan due to the unpredictability of lupus from the LupusQoL. Areas of physical health, particularly fatigue, were rated most frequently to be highly important and relevant to SLE patients. Domains unaddressed by existing SLE-speciﬁc measures identiﬁed were family, relationships, stigma and discrimination, and freedom. Similar subthemes of relationships could be identiﬁed from previous qualitative studies and are likely to be speciﬁc to SLE patients, including concerns about social isolation resulting from being misunderstood (4) and concerns about the image portrayed to others (8). Although themes regarding body image (33), attractiveness (8), and facial manifestations (4) were identiﬁed, stigma and discrimination was not conceptualized as a theme or domain in previous studies. The loss of freedom arising from the need to restrict one’s activities was also not reported as a theme. Certain subthemes from the family and stigma and discrimination domains could be more relevant in the Asian sociocultural context. A participant who has lived in a Western country compares the public stigma experienced in both contexts: “Of course they will stare at you, especially in Asia. . . . I mean, [in the Western country] they’ll have the courtesy, at least, not to stare. Of course there are some people that would stare. But I’m just saying that I feel that most of it is in Asia.” The concerns with childbearing difﬁculties may also be accentuated in the Asian culture, due to the importance of perpetuating the family name in the Chinese culture. A Chinese participant described the stress experienced when plans for a baby were put on hold due to a disease ﬂare: “It worries me, and I kind of feel stressed because my husband is an only son, so it adds a certain stress to me.” Dependence, burden to others, self-image, and symptoms (speciﬁc to SLE) are domains addressed by existing SLE-speciﬁc measures but not PROMIS Version 1.0 Item Banks. Notably, at least one subtheme from each domain is covered by either the LupusQoL or the SLEQOL. Participants also rated burden and self-image to be “highly relevant” as frequently as physical function. Therefore, until new measures are developed, using either instrument in combination with a generic measure provides additional HRQOL information relevant to SLE patients. Within the Asian population, the English and Chinese versions of the SLEQOL have been validated for use among SLE patients in Singapore (13,34). Upcoming PROMIS banks under development could address these domains in the future, including the Sexual Function bank and banks addressing aspects of social health (35). In a recent update of the Neuro-QoL, which focuses on HRQOL assessment in a variety of neurologic disorders and settings, stigma is now being addressed.
HRQOL Domains Important to Multiethnic Asian SLE Patients Items from the existing PROMIS Version 1.0 Item Banks generally addressed the corresponding domains well. Notably, the 3 areas of physical health rated most frequently by participants as highly important and relevant (fatigue, pain and other symptoms, and physical function) were addressed by the PROMIS Version 1.0 Item Banks. Therefore, PROMIS Version 1.0 Item Banks would serve as a useful core set of items for assessing HRQOL in SLE patients. However, new items need to be written for the identiﬁed gaps in some banks, owing to the peculiarities of SLE and its management. First, sleep disturbances experienced by SLE patients were often attributed to steroids, described by the unaddressed subtheme, “I do not feel the need to sleep.” Second, items (from the PROMIS Satisfaction with Social Participation [Discretionary Social Activities] bank) with the stems, “I am satisﬁed with my current level of/the amount of time I spend. . .” are more appropriate than items beginning with, “I am satisﬁed with my ability to. . ..” For SLE patients, activity limitation is not solely a result of physical inability to participate but also the need to refrain from certain activities, such as those in the sun. Third, family subthemes pertaining to childbearing and willingness to start a family are unique to SLE due to the predominantly female population, onset at a childbearing age, and fertility problems. Finally, the impact of SLE goes beyond the ability to work but includes job prospects, limiting one’s workload and stress, poorer motivation, and a negative impression to employers. Certain study limitations may preclude exhaustive coverage of topics for all subgroups of SLE patients, although it should be noted that as with much of qualitative research, focus group ﬁndings are not meant to be readily generalizable. Participants were ambulatory, recruited from outpatient sources, and not stratiﬁed by disease characteristics, such as duration of speciﬁc organ involvement. Although periods of hospitalizations and severe illness were recounted in the sessions, additional domains might have been elicited among SLE inpatients or the more severely ill. Our attempts to recruit inpatients for individual interviews were unsuccessful, as these patients were in an unsuitable state for participation. The views of non-English–speaking residents were not included, as only English-speaking participants were eligible for the study. However, 75% of the Singapore resident population ages 25– 65 years in 2010 is English literate (36). Furthermore, SLE patients are generally younger and many are English speaking. Therefore, our study results are generalizable to the majority of SLE patients in Singapore. Lastly, it is unknown whether topic saturation was achieved for male SLE patients, as only two male patients were recruited. Our study was conducted among patients in the Asian sociocultural context. Domains affected by ethnic-speciﬁc values, such as the importance of perpetuating the family name, could be similar throughout an ethnic group located across countries. However, the stigma faced by SLE patients living in Asia is likely to be speciﬁc to this sociocultural context. Although such an impact may not be relevant to other contexts, our ﬁndings contribute knowledge that ﬁndings from multiethnic SLE populations are not equivalent to patient perspectives directly obtained from each sociocultural context (4). Finally, PROMIS is an ongoing initiative with an evolving domain framework
907 (37); therefore, newly developed banks addressing the identiﬁed content gaps may be available by the time of study publication. Readers are advised to refer to the PROMIS web site (online at http://www.nihpromis.org) for the latest versions of the available item banks. In conclusion, 4 additional domains have been identiﬁed as content gaps in existing SLE-speciﬁc measures, speciﬁcally family, relationships, freedom, and stigma and discrimination. Two of these domains, family and stigma and discrimination, may be accentuated in the Asian sociocultural context. As stigma is a signiﬁcant factor in many but not all diseases, disease-speciﬁc item banks such as our study and the Neuro-QoL would provide additional value to PROMIS banks. As 40 subthemes from 14 domains identiﬁed in this study as important and relevant to SLE patients were addressed by the PROMIS Version 1.0 Item Banks (Physical Function, Pain, Fatigue, Sleep Disturbance, Sleep-Related Impairment, Anger, Anxiety, and Depression), PROMIS item banks have tremendous potential to serve as a core set of items for HRQOL assessment in SLE patients. Therefore, an SLE-speciﬁc item bank may be developed by writing new items to ﬁll the gaps in existing PROMIS item banks.
ACKNOWLEDGMENTS The authors thank the Lupus Association (Singapore) and Ms Connie Tse, Research Nurse at the Singapore General Hospital, for facilitating study recruitment, Ms Chew Ying Ying, the Department of Pharmacy, Faculty of Science, National University of Singapore, for study logistics, and Ms Ong Zhan Yuin and Ms Choo Qiu Yi for analysis of the transcripts. AUTHOR CONTRIBUTIONS All authors were involved in drafting the article or revising it critically for important intellectual content, and all authors approved the ﬁnal version to be published. Dr. Wee had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. Study conception and design. Thumboo, Cella, Cheung, Fong, Wee. Acquisition of data. Ow, Thumboo, Fong, Wee. Analysis and interpretation of data. Ow, Thumboo, Cella, Cheung, Fong, Wee.
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