Perceptions in 7700 patients with rheumatoid arthritis compared to their families and physicians

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Joint Bone Spine 74 (2007) 622e626 http://france.elsevier.com/direct/BONSOI/

Original article

Perceptions in 7700 patients with rheumatoid arthritis compared to their families and physicians Jacques Pouchot a,*, Jean-Marie Le Parc b, Laurence Queffelec c, Patrick Siche`re d, Alain Flinois e for the Association Franc¸aise des Polyarthritiques (AFP) a

Internal Medicine Department, European Georges Pompidou Teaching Hospital, 20 rue Leblanc, Paris 75015, France b Rheumatology Department, Ambroise Pare´ Teaching Hospital, Boulogne Billancourt, France c Association Franc¸aise des Polyarthritiques, Head Office, Paris, France d Rheumatology Clinic, Paris, France e Harris Me´dical International, Paris, France Received 15 June 2006; accepted 24 November 2006 Available online 19 July 2007

Abstract Objective: To compare perceptions of patients with rheumatoid arthritis (RA) to those of their families and usual physicians regarding pain and subjective experience of the disease. Methods: Questionnaires were mailed to patients listed in the files of a non-profit patient organization (Association Franc¸aise des Polyarthritiques). Each patient, one family member (or close friend), and the usual physician were each asked to complete a questionnaire. Concordance among replies made by patients, family/friends, and physicians was evaluated using the kappa coefficient. Results: Questionnaires were sent to 20,468 patients, among whom 7702 (38%) mailed back adequate data. The family member was usually the spouse (70%) and the usual physician a rheumatologist (68%). Joint pain was described by patients as variable (80%) and unpredictable (68%). Patients reported a need to push themselves (86%), frustration (86%), anxiety about possible disease progression (89%), and being prevented from making plans for the future (6%). A negative impact was reported on recreational activities (84%), work (56%), and family life and sexuality (51%). Concordance was excellent for pain severity (kappa > 0.90) and good for the main joint-pain characteristics and experience of the disease (kappa > 0.70), although family members tended to overestimate, and physicians to underestimate, the intensity of the pain. Conclusion: We found good overall agreement between perceptions of patients, their families, and their physicians, despite differences between these last two groups. Our qualitative analysis showed not only a major physical impact of the disease, but also marked negative psychosocial effects. Ó 2007 Elsevier Masson SAS. All rights reserved. Keywords: Rheumatoid arthritis; Joint pain; Disease experience; Concordance; Qualitative analysis

1. Introduction Rheumatoid arthritis (RA) is a chronic incapacitating disease that often causes joint destruction and functional impairment. RA is common, with an estimated prevalence in 2001 in France

* Corresponding author. Tel.: þ33 1 5609 3330; fax: þ33 1 5609 3816. E-mail address: [email protected] (J. Pouchot).

of 0.31% (95% confidence interval, 0.18e0.48%) overall, 0.51% in women, and 0.09% in men [1]. In addition to physical symptoms, severe alterations in psychological and social function develop early in the course of RA, as demonstrated by quality-of-life studies [2,3]. RA disrupts the life not only of the patients, but also of their families, a fact that has received little attention. Assistance and support from family and friends exert a strong influence on perceptions of the disease experience, most notably regarding

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J. Pouchot et al. / Joint Bone Spine 74 (2007) 622e626

psychological well-being and coping strategies [4,5]. The spouse is usually the main source of emotional and material support for the patient with a chronic disease. A prerequisite to providing optimal support is a perception of the disease experience that is largely consonant with that of the patient. Discrepancies in perceptions, which may be considerable, are associated with a negative impact on patient well-being, leading to a higher rate of depression [6e9]. Although patients with RA may report that their family or physicians do not understand, differences in perceptions between patients and others have not been extensively studied [10]. Discrepancies in perceptions are probably smaller regarding observable factors (e.g., functional abilities) than regarding the emotional impact of the disease. A close match between the patient’s perceptions and those of the family and physician should help to organize appropriate support, optimize disease management, and ultimately improve quality of life. The objective of this study was to compare perceptions of the disease by patients, family/friends, and physicians. 2. Methods We conducted a cross-sectional mail questionnaire survey with the help of the non-profit patient organization Association Franc¸aise des Polyarthritiques (AFP). The questionnaires were developed during meetings of patients with RA, rheumatologists, other physicians, and methodologists. In a preliminary qualitative study, semi-structured interviews of RA patients generated data on perceptions of the disease experience from symptom onset to the day of the interview and on past and present difficulties related to the disease. A steering committee composed of AFP representatives and rheumatologists closely involved in RA management validated the survey tools and accompanying documents. Harris Medical International (Paris, France) developed the survey tools, implemented and monitored the survey, and conducted the statistical analyses. In 2002, patient questionnaires were mailed to the 20,468 patients with RA who were on AFP files. Each patient received a questionnaire, a letter explaining the study, and a prepaid envelope for returning the completed questionnaire. AFP files include members and non-members. The limited data available in the AFP files precluded quota sampling. Given the modest response rate usually associated with mail surveys, we therefore sent questionnaires to all the patients in the AFP files. Each patient was asked to supply contact information for one family member or close friend and for the physician most closely involved in the management of the disease. Reminders were not sent to non-responders. This phase of the survey lasted 4 months. Physician questionnaires were mailed upon receipt of the patient questionnaires. Concomitantly, family members (or friends) were interviewed over the telephone. The patient questionnaire comprised 35 items organized in seven sections: patient-reported diagnosis of RA (as evaluation of American College of Rheumatology criteria was not feasible), follow-up, information supplied to the patient about the disease, pain, perceived experience of the disease, activity

623

restrictions, and help received. The patients also completed the validated French translation of the Health Assessment Questionnaire (HAQ) [11], which served to evaluate physical function and functional disability; the HAQ score can range from 0 (no disability) to 3 (greatest possible disability), The questionnaires used for the close family member or friend and for the main physician were modeled on the patient questionnaire sections on pain and perceptions of the disease experience. The statistical analysis of patient questionnaire data involved computation of means, medians, standard deviations, and ranges for quantitative variables; and numbers and percentages for qualitative variables. Student’s test was used to compare quantitative data and the chi-square test qualitative data. Concordance between patient data and family or physician data were evaluated based on the kappa coefficient [12]. A kappa coefficient value of 1 indicates complete disagreement, þ1 complete agreement, and 0 independence between raters. Kappa coefficient values greater than 0.8 are considered to indicate excellent agreement, values of 0.61e0.8 good agreement, values of 0.41e0.6 moderate agreement, values between 0.21 and 0.4 mediocre agreement, and values smaller than 0.21 poor agreement [13]. 3. Results 3.1. Description of the population Of the 20,468 patients to whom questionnaires were sent, 7627 mailed back assessable data. In addition, 75 patients volunteered to participate in the study. Thus, the patient sample comprised 7702 individuals (7702/20,468, 38%). Table 1 reports the main patient characteristics. Mean (SD) disease duration since symptom onset was 15.5  11.4 years and mean patient-reported time from symptom onset to diagnosis was 2 years. Mean HAQ score was 1.11  0.73 overall, 1.16  0.73 in women, and 0.91  0.70 in men. These values were similar to those obtained by Wolfe et al. [14] in 2491 patients who were representative of the RA population in the US. The HAQ score correlated with age, disease duration, and patient-reported diagnostic delay. No data on rheumatoid factor were collected. At the time of the survey, 89% of patients were taking disease-modifying anti-rheumatic drugs, of which the most widely used was methotrexate (45%). Nearly half the patients were taking glucocorticoid therapy, whereas only 6% were on TNFa antagonist therapy in this 2002 survey. AFP members contributed 43% (3304/7702) of the sample; compared to non-members, they were older (58.7 years vs. 55.8 years, P < 0.0001), had longer disease durations (16.2 years vs. 14.9 years, P < 0.0002), and had higher HAQ scores (1.18 vs. 1.05, P < 0.0001). Of the 7702 respondents, 4002 (52%) supplied contact information for a close family member or friend, and 3008 (75%) of these individuals were interviewed over the phone. Spouses contributed 70% of family members and parents 11%. Contact information for the main physician was supplied by 5564 (72%) respondents, and 1918 (1918/5564, 35%) physicians mailed

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Table 1 Main characteristics of the 7702 patients with rheumatoid arthritis included in the study Characteristics Mean age (years) Women Disease duration (years since first symptoms reported by the patient) HAQ disability scorea Married or living with a partner One or more children Occupational status Retired Paid employment No occupation Unemployed Incapacitated

Mean (SD)

Number (%)

57 (13.8) 6252 (81%) 15.5 (11.4) 1.11 (0.73) 5351 (69%) 6185 (80%)

Treatments No DMARDb Methotrexate Hydroxychloroquine Sulfasalazine Leflunomide Gold TNFa antagonists Glucocorticoids

3185 2204 638 259 1821

(41%) (29%) (8%) (3%) (24%)

833 3444 1210 798 793 430 454 4099

(11%) (45%) (16%) (10%) (10%) (6%) (6%) (53%)

a HAQ: Health Assessment Questionnaire for disability. The score can range from 0 (no disability) to 3 (greatest possible disability). b DMARD: disease-modifying antirheumatic drugs.

back completed questionnaires. Of the 1918 physician respondents, 1306 (68%) were rheumatologists and 560 (29%) were general practitioners.

Moderate or worse pain was reported by 75% of respondents during the last week and 84% during the last year (Table 2). Pain severity correlated with the HAQ score (r ¼ 0.45 with pain in the last week, P < 0.001). Patients were asked to rate seven pain characteristics on a four-level verbal scale from ‘‘strongly agree’’ to ‘‘strongly disagree’’ (Table 2). The main reported characteristics were variability (80% of ‘‘strongly agree’’ or ‘‘agree’’ responses), unpredictability (68%), and major interference with paid work or domestic chores (67%). Underestimation of pain by the spouse and physician was reported by only 23% and 14% of patients. In contrast, 38% of patients reported underestimation of pain severity by other family members or friends. 3.3. Perceptions of the disease experience We investigated the impact of RA on psychological wellbeing during the last year and on activities and personal projects at any time. The four-level scale used for psychological well-being and the three-level scale for activities are shown in Table 3. Negative feelings were reported more often than positive feelings. Most patients reported having to push themselves (89%), frustration at being unable to do things (86%), anxiety about future disease progression (82%), depressive symptoms (75%) and an inability to make plans for the future (67%). The replies to items on activities showed that RA negatively affected recreational activities (84%), work-related activities (56%), sexual activities (51%), family life (51%), and intimate relationships (44%). 3.4. Concordance between patients and family/friends or physicians

3.2. Joint pain Joint pain was evaluated using two timeframes: the last week and the last year. Pain severity was assessed on a five-level verbal scale (none, mild, moderate, severe, and very severe).

Concordance was investigated for pain severity over the last year, main characteristics of the pain, and selected perceptions

Table 2 Joint pain severity and characteristics in the 7702 patients with rheumatoid arthritis Severity of joint pain

Very severe

Over the last 8 days Over the last year

5% (353) 6% (468)

Severe

Moderate

Mild

No pain

23% (1777) 31% (2422)

47% (3595) 47% (3590)

18% (1383) 12% (936)

6% (461) 2% (130)

Characteristics of joint pain

Strongly agree

Agree

Disagree

Strongly disagree

Continuous Varies from day to day Brought on by activities Unpredictable Responds poorly to medications Major impediment to activities Severely alters relationships with family, friends, and acquaintances

27% 48% 25% 42% 19% 39% 19%

18% 32% 27% 26% 31% 28% 21%

12% 3% 11% 6% 19% 10% 18%

12% 3% 9% 4% 12% 6% 19%

Perception that the joint pain is underestimated by

Strongly agree

Agree

Disagree

Strongly disagree

The spouse The physician The family (except spouse) Friends Co-workers

10% 5% 17% 15% 9%

13% 9% 21% 23% 9%

13% 18% 16% 16% 6%

27% 41% 25% 20% 8%

(2066) (3691) (1952) (3248) (1488) (2993) (1445)

(798) (363) (1276) (1156) (691)

(1404) (2494) (2109) (2024) (2366) (2139) (1655)

(968) (728) (1605) (1802) (711)

(897) (205) (830) (491) (1475) (778) (1 394)

(993) (1424) (1221) (1243) (489)

(895) (193) (679) (333) (901) (427) (1 501)

(2091) (3190) (1902) (1570) (601)

J. Pouchot et al. / Joint Bone Spine 74 (2007) 622e626

625

Table 3 Perceptions of the disease experience by 7702 patients with rheumatoid arthritis Psychological well-being

Very often

Often

Occasionally

Never

I have to push myself to do things that used to be easy. I am doing better about rolling with the punches. I feel frustrated because my disease prevents me from doing things I want to do. I feel annoyed because my family/friends fuss over me. I am not as hard on myself, because I accept the limitations due to my disease. My disease puts me in embarrassing or humiliating situations. I suffer anxiety because I am afraid my disease will get worse. I am doing better about listening to others. I feel sad, depressed, and low. I depend on others. I feel emotionally stronger. I feel rejected and shut out. My disease prevents me from making plans for the future

34% 21% 39% 6% 17% 7% 29% 22% 19% 11% 15% 4% 19%

30% 32% 24% 9% 29% 8% 20% 35% 17% 16% 23% 4% 18%

25% 24% 23% 30% 30% 28% 32% 23% 39% 36% 28% 16% 31%

4% 6% 6% 40% 12% 42% 11% 5% 15% 23% 17% 61% 24%

(2625) (1625) (3016) (465) (1 271) (503) (2242) (1695) (1469) (849) (1145) (281) (1452)

(2298) (2484) (1867) (681) (2221) (652) (1571) (2731) (1286) (1245) (1793) (318) (1390)

Activities and projects: RA impacts negatively on

Yes, undoubtedly

Yes, probably

No

Recreational activities Work-related activities Family life Sexual activities Intimate relationships Relationships with friends Plans for having children

59% 41% 21% 23% 19% 12% 9%

25% 15% 30% 28% 25% 26% 6%

9% 24% 35% 33% 37% 46% 52%

of the disease experience. For sensitive items, we confined the concordance evaluation to patients and family/friends. Kappa coefficients were greater than 0.90 for pain severity, indicating excellent concordance between patients and family/friends or physicians. Kappa coefficients were somewhat lower, although always greater than 0.70, for pain characteristics and perceptions of the disease experience (Table 4). Family/friends often tended to overestimate pain severity and characteristics and to underestimate negative effects of RA on the patient’s life. Physicians, on the contrary, tended to underestimate pain severity and characteristics (Table 4). No differences in concordance were found according to disease duration (<5 or 5 years) (data not shown).

4. Discussion The results of this large-scale survey establish that most RA patients experience at least moderate joint pain that is both variable and unpredictable. In addition, patients perceive a negative impact of the disease on most of the areas of their lives. In patients with chronic diseases such as RA, support from the usual physician, spouse, and other members of the close circle of family and friends improves acceptance of the disease, quality of life, and well-being. Conceivably, these benefits might translate into better disease outcomes. Concordance, as evaluated using the kappa coefficient, was high overall in our study. However, item-by-item analyses revealed common overestimation of pain and its main characteristics by family and friends, contrasting with underestimation of the negative perceptions of the disease by the patient. Riemsma et al. [10] reported results of similar nature and greater magnitude: among spouses, 57% overestimated and 23% underestimated pain severity, while 40% overestimated and 32% underestimated

(4572) (3132) (1648) (1750) (1478) (943) (722)

(1907) (1169) (2332) (2156) (1947) (2010) (478)

(1937) (1838) (1756) (2275) (2303) (2130) (2468) (1755) (3009) (2783) (2167) (1221) (2355)

(319) (439) (458) (3045) (920) (3268) (816) (396) (1189) (1785) (1327) (4665) (1819)

(684) (1869) (2661) (2480) (2846) (3552) (4024)

functional impairment. In our study, physicians tended to underestimate pain severity and characteristics. Reasons for differences in perceptions between patients and family/friends were not investigated in our study. Early in the disease, family and friends may lack the medical information needed to grasp the implications of RA. Later on, burn-out may impair their ability to understand the patient’s experience. A study designed to investigate the causes of disagreement would be needed to determine whether these speculations hold merit. The methodological limitations of our study should be borne in mind when interpreting the results. Identification of the study patients from the files of a patient organization (Association Franc¸aise des Polyarthritiques) may have introduced selection bias. Overall, the disease was fairly severe (HAQ score, 1.11) and long-standing. Conceivably, a study confined to patients with early-onset RA might show greater levels of disagreement, although an analysis in a small subgroup of patients with early RA in our sample does not support this possibility (data not shown). Because we surveyed the patients, we were not able to evaluate ACR criteria for RA. As a result, we cannot exclude that some patients did not have RA. However, several findings indicate that any misclassification was minimal: the patients were identified through an organization for RA patients, 90% of patients were taking disease modifying antirheumatic drugs widely used to treat RA, and all the physicians who completed the questionnaire (35% of the sample) confirmed the diagnosis of RA. Response bias may have led to overestimation of the level of agreement: patients may have been more likely to supply contact information for family/friends and physicians whom they believed understood their disease experience. Only 52% of patients supplied contact information for a family member or friend, who was the spouse in 70% of cases. In the few

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J. Pouchot et al. / Joint Bone Spine 74 (2007) 622e626

Table 4 Concordance between perceptions by patients and by family/friends or physicians Family/friends

Physicians

Underestimation Complete agreement Joint pain Intensity over the last year Characteristics Continuous Varies from day to day Unpredictable Responds poorly to medications Major impediment to activities Severely alters relationships with family, friends, and acquaintances Activities: negative impact of the disease on Recreational activities Work-related activities Family life Sexual activities Intimate relationships Relationships with friends Plans for having children

Overestimation Kappa Underestimation Complete Overestimation Kappa agreement

16% (442)

46% (1265) 38% (1 062)

0.92

33% (601)

47% (852) 20% (372)

0.93

28% 22% 33% 27% 19% 27%

(577) (540) (775) (657) (456) (640)

38% 49% 41% 34% 47% 34%

(780) (1213) (969) (818) (1109) (798)

34% 30% 26% 39% 34% 39%

(713) (734) (627) (941) (819) (924)

0.78 0.82 0.76 0.77 0.82 0.76

45% 42% 63% 50% 43% 31%

32% 40% 25% 31% 39% 33%

0.75 0.83 0.72 0.79 0.82 0.78

26% 31% 36% 34% 31% 28% 13%

(702) (635) (907) (747) (712) (693) (229)

56% 55% 46% 50% 52% 55% 74%

(1505) (1125) (1164) (1117) (1197) (1362) (1273)

18% 13% 19% 16% 17% 18% 13%

(484) (271) (483) (359) (380) (437) (227)

0.72 0.71 0.71 0.74 0.72 0.73 0.71

(532) (660) (860) (724) (653) (423)

(387) (635) (342) (445) (584) (455)

23% 18% 12% 20% 18% 35%

(274) (277) (161) (288) (275) (481)

For highly sensitive items, concordance was evaluated with family/friends but not with physicians.

studies comparing responses from patients with non-rheumatic diseases and their physicians or family members, agreement was usually poor, indicating a need to rely on patient data when evaluating pain or quality of life [15e17]. In patients with chronic diseases such as RA, documenting e and identifying the reasons for e discrepancies in perceptions may help to improve the level of understanding among those involved, thereby optimizing the support provided by the circle of family and friends, as well as by physicians. Emotional, material, and informational support plays a crucial role in the course of RA. Regrettably, no data on this issue have been obtained in France. Evaluations conducted using validated instruments would provide the information needed to design programs for optimizing support to patients with RA. Acknowledgments We are grateful to Schering Plough Inc. for supporting this study. References [1] Guillemin F, Saraux A, Guggenbuhl P, Roux CH, Fardellone P, Le Bihan E, et al. Prevalence of rheumatoid arthritis in France: 2001. Ann Rheum Dis 2005;64:1427e30. [2] Rupp I, Boshuizen HC, Jacobi CE, Dinant HJ, van den Bos GA. Impact of fatigue on health-related quality of life in rheumatoid arthritis. Arthritis Care Res 2004;51:578e85. [3] Suurmeijer TP, Waltz M, Moum T, Guillemin F, van Sonderen FL, Brianc¸on S, et al. Quality of life profiles in the first years of rheumatoid arthritis: results from the EURIDISS longitudinal study. Arthritis Care Res 2001;45:111e21. [4] Doeglas D, Suurmeijer T, Krol B, Sanderman R, van Rijswijk M, van Leeuwen M. Social support, social disability, and psychological wellbeing in rheumatoid arthritis. Arthritis Care Res 1994;7:10e5.

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