Long-term follow-up of 246 adults with juvenile idiopathic arthritis: predictive factors for mood and pain

J. C. Packham, M. A. Hall and T. J. Pimm1

Oxford Regional Paediatric Rheumatology Unit, Wexham Park Hospital, Slough SL2 4HL and
1 Oxford Regional Rheumatic Diseases Research Centre, Stoke Mandeville Hospital, Aylesbury HP21 8AL, UK

Abstract

Objective. To examine the predictive factors for anxiety, depression and pain in adults with juvenile idiopathic arthritis (JIA).

Patients and methods. Two hundred and forty-six adults identified with long-standing JIA had an average disease duration of 28.3 yr. Candidate factors potentially predictive for pain, anxiety and depression were assessed by multiple regression analysis.

Results. Of the patients, 31.6% were anxious, 5.2% were depressed, and 21.1% had previously suffered from depression. The percentage of the variance accounted for by other variables was 78.8 for anxiety variance and 54.5 for depression, but there was little influence from physical disease-related factors. Severe pain, measured on a visual analogue scale, occurred in 32.9% of patients, and 22.8% had poor perceived control over their pain. Function, coping strategies, pain self-efficacy, inflammation and previous depression could predict 39.6% of the variance in pain.

Conclusions. Comparing adults with children, disease activity and control over pain remain predictors of pain but become less important than disability and coping strategies.

KEY WORDS: Juvenile idiopathic arthritis, Pain, Anxiety, Depression, Mood, Self-efficacy, Coping strategies, Long-term follow-up.

There are features of juvenile idiopathic arthritis (JIA) that suggest that sufferers may have a higher risk of psychological complications, such as pain, disability, physical deformity and onset in childhood. A number of studies have shown that psychological problems, particularly depression, are higher in people with inflammatory arthritis compared with the general population, e.g. the prevalence in rheumatoid arthritis is 14–46% [14]. The major psychological difference between adult-onset inflammatory arthritis and JIA is that coping strategies are not fully developed in childhood, so that adolescence has to be negotiated with a chronic disease. This may affect the long-term psychological health of the individual and their ability to cope with disability in adulthood.

The long-term psychological implications for adults with JIA have not been studied in as much depth as in adults with rheumatoid arthritis [59]. David et al. [5] reported clinical depression in 21% of 43 adults with polyarticular JIA, the rate increasing with the degree of disability. Anxious preoccupation with disease rose with worsening physical function and disease activity. Anxious and helpless responses were seen more commonly in patients whose arthritis started in adolescence, possibly because adolescents have less time to adapt and develop alternative coping strategies compared with those with arthritis from early childhood. Aasland et al. [7] found that 17% of 52 adult JIA patients had a psychiatric diagnosis, often anxiety, but none had a depressive disorder. Peterson et al. [8] suggested that ‘JRA cases were not emotionally impaired and were able to perform social activities similar to controls’. Wirrell et al. [9] found that 64 adults with JIA were within the normal range for emotional well-being, pain and energy.

Pain is a major symptom in JIA and may detrimentally affect the health of patients. Active inflammatory arthritis is accompanied by increased pain, worsening fatigue and morning stiffness. Despite studies assessing pain in rheumatoid arthritis and osteoarthritis and in children with JIA, pain in adults with JIA has been understudied. Early studies suggesting that children with JIA experience less pain than rheumatoid adults [10, 11] are now thought to be misleading. Recent use of age-appropriate measures of pain show that pain in children with JIA is common [12, 13]. Sherry et al. [14] found that 97% of children with polyarticular JIA at a routine paediatric rheumatology clinic reported some degree of pain. Schanberg et al. [13] found that 25% of JIA patients were in the middle to high range on pain scales despite regular attendance at a specialist centre, where analgesic control should have been optimal.

Most pharmacological strategies reduce pain by analgesics and control of active inflammation. However, chronic pain is a biological and psychological phenomenon. Multiple regression models suggest that disease severity does not strongly relate to pain, joint inflammation accounting for 10% of pain variance [15]. Thompson et al. [16] found that modest amounts of variance in JIA pain were accounted for by arthritis subtype (8%) and disease activity (1%).

Multi-dimensional assessments (demographic, medical, psychological and coping strategy variables) predict pain variance more effectively. Schanberg et al. [13] predicted just over 50% of the variance in pain, the majority explained by disease activity and coping strategies. Children and adults who feel their self-efficacy over pain is strong have fewer tendencies to catastrophize (to think negatively or destructively) [17] and to have lower pain intensity scores. Psychosocial variables, such as coping strategies and self-efficacy, are therefore important when considering the treatment of pain in children with JIA.

Children and adults with persistent pain develop strategies to cope with their pain. Passive coping and catastrophizing have been consistently linked to higher pain levels and poor functional outcome. Other strategies (calming self-statements, distraction, etc.) have been shown to produce lower levels of pain. Few studies have examined the efficacy of pain coping strategies in JIA.

Newman and Revenson [18] classified 158 adult rheumatoid arthritis patients into four groups on the basis of their overall pattern of coping strategy, using the London Coping with Rheumatoid Arthritis Scale (Table 1Go). Reports of pain and stiffness were significantly lower for group 3, the ‘open copers’; this group had less physical disability and higher levels of psychological well-being. This suggests that different coping strategies impact on reporting of symptoms and disability.


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TABLE 1. Coping strategy classification

 
Predictive factors influencing pain levels are likely to change as patients enter adulthood. As the duration of these patients' disease course lengthens, levels of disability and joint destruction worsen. A combination of functional limitation, reduced support following independence and increased demand can limit in adulthood the level of independence achieved in adolescence. As the learning period of childhood and adolescence is left behind, coping skills develop and previous experiences (both good and bad) affect patients' psychosocial state.

This is the largest study describing predictive factors for anxiety, depression and pain in JIA adults. Mood and pain have a detrimental effect on an individual's quality of life and their ability to cope with disability. A better understanding of the factors that predict mood and pain may allow both patients and health professionals to address these problems more effectively.

Methods

Out of 259 adults meeting the ILAR criteria for JIA [19] who were identified, 246 (95%) attended for an interview, clinical examination and notes review by the same rheumatologist (JCP). Two hundred and thirty-one (89.2%) returned a comprehensive functional and psychosocial self-assessment questionnaire.

Mood was measured with the Hospital Anxiety and Depression (HAD) scale [20], a 14-point scale which contains seven questions on anxiety symptoms and seven on depressive symptoms. The HAD scale was specifically designed for use in patients with physical illness, the depression scale being constructed to exclude somatic items on fatigue or sleep disturbance (often caused by physical illness) and emphasize anhedonia (loss of pleasure). The age at onset of previous mood imbalance was noted, defined by the presence of a psychiatric diagnosis of anxiety or depression, the prescription of antidepressant medication or a parasuicide attempt. The patient's perception of the effect of arthritis on mood was measured using the Disease Repercussion Profile (DRP) [21].

Patients rated pain levels using a 100-mm horizontal visual analogue scale (VAS). The end-points of the scale were ‘no pain’ (zero) and ‘most severe pain’ (100). The Arthritis Self-Efficacy Scale [22] was used to indicate the patient's perceived level of control over their pain. Medication use, particularly non-steroidal anti-inflammatory drugs (NSAIDs) and simple analgesics, indicated those patients requiring continuing analgesic support.

Candidate predictive factors with the potential to affect anxiety, depression and pain were reviewed as follows. Patient-related physical demography—gender, weight, height, physical activity, mobility and growth defects; patient-related social demography—education, marital state, offspring, employment, discrimination, housing and disability benefits; disease demography—age at onset, disease duration, JIA subset, medication use, JIA-associated diseases (uveitis, osteoporosis and amyloid) and medical and surgical history; physical assessment—joint inflammation (Thompson–Kirwan scale) [23], range of joint movement, Steinbrocker score [24] and UK-validated version of the ‘Health Assessment Questionnaire’ (HAQ) [25]; laboratory assessment for current disease activity—C-reactive protein (CRP), erythrocyte sedimentation rate (ESR), haemoglobin, white cell count, platelets and IgG; coping strategies— London Coping with Rheumatoid Arthritis Scale [18] and the catastrophizing subset of the Coping Strategies Questionnaire [26]; cognition—Arthritis Self-Efficacy Scale [22] (which measures a patient's belief that they control certain symptoms or can perform certain tasks; this scale has three subsets assessing the areas of pain, function, and other symptoms); social support—Sarason's Short Form Social Support Questionnaire [27]; perceived handicap—DRP, perceived impact of arthritis on a patient's lifestyle, including physical activity, social activity, employment and finances, relationships with family, friends and partners, appearance or body image, and emotional state.

Correlation between mood, pain and other variables was assessed using Spearman's correlation coefficient (two-tailed). Candidate predictive factors were initially assessed using a matrix of Spearman's correlation coefficients. Those factors with significant correlation coefficients (P<0.05) were entered into a multiple linear regression analysis, testing for variables capable of predicting anxiety or depression measured with the HAD scale and pain measured with the pain VAS.

Results

Of the 246 individuals who were interviewed and examined, 70 (28%) were men and 176 (72%) women. The mean age at review was 35.4 yr (range 18–71) and the mean duration of arthritis was 28.3 yr (range 9–71). The mean age at disease onset was 7.1 yr (range 0.8–15.9). The frequency of JIA subsets in the study group compared with a paediatric JIA population was skewed towards those subsets with a poor functional outcome [28].

The percentage of patients with a high anxiety level (HAD score>8) was 31.6 but only 5.2% had high levels of depression (HAD score >8). Patients with systemic-onset JIA had significantly higher levels of anxiety (41.7%, P<0.05) and depression (10.7%, P<0.05) and patients with oligoarticular JIA had lower levels of anxiety (7.7%, P<0.05) compared with patients in the other JIA subsets.

Depression was most commonly seen when the age at onset of JIA was between 6 and 12 yr (11.1%, P<0.01) compared with early (2.7%) or late (0%) onset JIA. Those patients in the late-onset group over 12 yr had the highest risk of developing anxiety-related problems (41.5%, P<0.05), compared with the middle (29.6%) and early (28.7%) groups.

The percentage of patients who had experienced significant depression in the past was 21.1. Most commonly, the first episode of depression tended to be between the ages of 15 and 25 yr (38.5%), followed by 26–35 yr (22.9%), 36–45 yr (17.3%) and 46–55 yr (3.8%). Retrospective analysis of anxiety was felt likely to be inaccurate, due to under-reporting and multiple uses of anxiolytics.

The DRP assesses the impact a patient feels that their disease has had on different aspects of their life; 37.9% of patients felt that their emotional state had been negatively affected by JIA, with a mean score of 2.8/10, and 26% of patients felt that their arthritis exerted a severe detrimental effect (score of 8/10 or higher) on their emotional state.

Only 7% of patients were pain-free, 35.1% scored 1–25 on the pain VAS, 25% scored 26–50 and 32.9% over 50. The mean score overall was 37. Oligoarticular patients experienced less pain (mean 20.5, P<0.01) and systemic patients experienced more pain (mean 47.9, P<0.05). The percentage of patients who still used NSAIDs was 72.4; 30.1% used simple analgesics and overall 79.7% required some form of analgesic support. There was a significant correlation between pain intensity and analgesic use (P<0.001).

Control over pain, measured with the Arthritis Self-Efficacy Scale, was complete/good in 32% of patients, moderate in 45.2% and poor/very poor in 22.8%. The mean self-efficacy score was 65/100. The rheumatoid factor-negative polyarticular JIA patients perceived more control over pain (mean 69.9, P<0.05) than other subsets. A similar trend in oligoarticular and psoriatic JIA groups did not reach significance due to the small number of patients in each group. Systemic patients experienced less control over pain (mean 60.4, P<0.05). There was an inverse correlation between pain VAS and pain self-efficacy score (P<0.001). Pain self-efficacy was not associated with CRP and only weakly related to clinical inflammation (P<0.005).

The best model of forward stepwise multiple regression analysis testing (Table 2Go) identified eight variables that independently made a significant contribution (78.8%) to the variation in patient's level of anxiety. Four variables accounted for 54.5% of the variation in depression levels. Six variables accounted for 39.6% of the variance in pain levels. Three variables accounted for 24.3% of the variance in control of pain (self-efficacy). A separate regression analysis on the four individual coping strategies suggested that they were relatively independent of other factors in view of poor predictability, varying from just 1.4% of variation for passive coping strategies to 11.3% for open coping strategies.


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TABLE 2. Predictive factors for anxiety, depression, pain and pain self-efficacy in adults with JIA

 

Discussion

The level of depression in this study group (5.2%) was lower than that of the general population and similar chronically disabled groups. Turner and McLean [29] screened 22 000 adults and identified 731 (6.7%) with physical disability. Of these, 37% were depressed and 46% anxious, compared with 12 and 18% respectively of the matched population. Even taking into consideration the different measures of mood used in this study, the level of depression in our study group appears to be relatively low.

Previous depression was common, often occurring in the late teens or early twenties. At this age, individuals tend to leave home and seek independence; consequently this is the time when coping techniques are finalized and put under the most strain. Depressive episodes become less common in later life, suggesting that experience enables patients to learn to cope with their disease more effectively. This supports the hypothesis suggested by Timko et al. [30] that psychosocial adjustments continue with time as an individual adapts to their disease. Although previous depression continues to be an indicator of anxiety later in life (P<0.005), it was not associated with continued depression at follow-up.

As previously reported by David et al. [5] and Aasland et al. [7], the levels of anxiety found in adults with JIA were well above those seen in the general population. Similar levels of anxiety are found in other well-controlled large studies of adults with disability related to other diseases [29]. In patients in whom arthritis does have an effect on emotional well-being, this effect is unlikely to be perceived as small, and in this group there is a strong influence of arthritis on mood. Patients with systemic-onset JIA had higher scores for both anxiety and depression, which may be related in part to the high levels of physical disability and dependency seen in this subset. Patients with oligoarticular arthritis had significantly less anxiety than other subsets, reflecting the less severe course and effects of JIA in this group.

Pain intensity in adults with JIA (mean 37.9) was higher than in reports for children with JIA (mean 16.3–29.7) [13, 31]. This may be related to a combination of increasing disease-related damage over time and the severity of disease in the group studied. Adults may also be more prepared to express pain on a self-report questionnaire.

Physical disease factors had little influence on the presence of a poor psychological state. All of the markers for depression had a psychological base, and clinical inflammation accounted for only 3.6% out of a predictive total of 78.8% for anxiety. Two important causes of anxiety and depression are a lack of satisfaction with levels of social support and poor body image. In anxiety and depression the most important predictive factor was self-efficacy, a patient's belief that they can achieve a specific behaviour or control a specific symptom. This measure may indicate either less predictability of symptoms in these patients or a difficulty coping with similar levels of symptoms experienced by other patients. Self-management courses, with specific techniques being learnt and practised, not only improve self-efficacy but also benefit patients' health outcomes [22]Go.

Forty per cent of the variation in pain could be accounted for by six independent variables lying within two areas. There was a direct physical influence on pain from physical function and joint inflammation, and a psychological influence on pain from self-efficacy, previous depression and coping strategies. This contrasts with the predictive factors for pain in the paediatric JIA population. Schanberg et al. [13] showed that in paediatric JIA, disease activity, present in 84% of patients, predicted 28% of the variation in pain levels (Table 3Go). However, in adults the role of disease activity as a predictor for pain becomes less important, accounting for just 2.2% of the variation in pain, despite detectable active joint inflammation present in almost half of the study population. In adult JIA, physical function and disability, which deteriorate with increasing length of disease, take precedence, the HAQ score predicting 18.3% of the variability in pain.


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TABLE 3. Predictive factors for pain in adult and paediatric JIA

 
It appears that, as a patient enters adulthood, pain coping strategies become a more important predictor of pain. Two coping strategies (‘denial’ and ‘dependence’) are detrimental to pain intensity. In both strategies the disease and its related symptoms are not confronted. In contrast, the presence of an ‘open’ coping strategy, in which disease is actively confronted, correlates well with lower levels of pain intensity (P<0.005) and disability (P<0.01). An open coping strategy is also protective against the onset of anxiety, whilst catastrophizing (destructive or negative thinking) increases the chance of developing an anxiety state.

The age at onset of disease may have an effect later in life on the effectiveness of learned coping strategies to avoid anxiety or depression. The mid-onset group was more depressed and the late-onset group more anxious compared with other age groups, despite similar disease demographics in all the groups. The apparent benefit to psychological health in the early-onset group may be related to a lack of sufficient cognitive development [32] to comprehend the potential effects of arthritis. In the mid- and late-onset groups there may be a more pronounced effect of loss due to JIA on the development of self-identity and self-confidence. The effect of age at disease onset on coping strategies and their subsequent development warrants prospective study in children and adolescents with JIA.

Present mood did not appear to have any predictive value for pain intensity. Although there was a strong correlation between pain intensity and both depression (P<0.001) and anxiety (P<0.001), this is explained by other factors in the linear regression analysis. It is the presence of previous depression that predicts pain. As with the other psychological predictive factors, such as coping strategies and perceptions of pain control, this may be viewed as an indicator of how well an individual has learnt to manage their disease over time.

Psychological variables, rather than acute effects of inflammatory disease, explain the majority of the variance in depression and anxiety in adults with JIA, and both physical and psychological factors influence pain. A holistic approach is necessary, in which not only the patients' physical needs are met but also their psychological needs. Effective psychological strategies enabling patients to cope with the stressors of chronic arthritis may be just as effective as reaching for the prescription pad.

Acknowledgments

This research was supported by a grant from the Arthritis Research Campaign.

Notes

Correspondence to: J. C. Packham, Staffordshire Rheumatology Centre, Haywood Hospital, High Lane, Burslem, Stoke-on-Trent ST6 7AG, UK. Back

References

  1. Creed F. Psychological disorders in rheumatoid arthritis: a growing consensus? Ann Rheum Dis1990;49:808–12.[Abstract]
  2. Baum J. A review of the psychological aspects of rheumatic diseases. Semin Arthritis Rheum1982;11:352–61.[ISI][Medline]
  3. Bishop D, Green A, Cantor S, Torresin W. Depression, anxiety and rheumatoid arthritis activity. Clin Exp Rheumatol1987;5:147–50.[ISI][Medline]
  4. Frank FG, Beck NC, Parker JC et al. Depression in rheumatoid arthritis. J Rheumatol1988;15:920–5.[ISI][Medline]
  5. David J, Cooper C, Hickey L et al. The functional and psychological outcomes of juvenile chronic arthritis in young adulthood. Br J Rheumatol1994;33:876–81.[ISI][Medline]
  6. Doherty E, Rooney M, Coneroy R, Bresnithan B. Health status of functionally independent young adults with chronic arthritis since childhood. J Orthop Rheumatol1988;1:51–8.
  7. Aasland A, Flato B, Vondivik LH. Psychosocial outcome in juvenile chronic arthritis: a nine year follow-up. Clin Exp Rheumatol1997;15:561–8.[ISI][Medline]
  8. Peterson LS, Mason T, Nelson AM, O'Fallon WM, Gabriel SE. Psychosocial outcomes and health status of adults who have had juvenile rheumatoid arthritis. Arthritis Rheum1997;49:2235–40.
  9. Wirrell E, Lang B, Canfield C. Social outcomes in young adults with juvenile arthritis: implications for the development of transition clinics. Arthritis Rheum1995;38:S188.
  10. Laaksonen A, Laine V. A comparative study of joint pain in adult and juvenile rheumatoid arthritis. Ann Rheum Dis1961;20:386–7.[ISI][Medline]
  11. Scott PJ, Ansell BM, Huskisson EC. Measurement of pain in juvenile chronic polyarthritis. Ann Rheum Dis1977;36:186–7.[Abstract]
  12. Benestad B, Vinge O, Veierod MB et al. Quantitative and qualitative assessments of pain in children with juvenile chronic arthritis based on the Norwegian version of the Pediatric Pain Questionnaire. Scand J Rheumatol1996;25:293–9.[ISI][Medline]
  13. Schanberg LE, Lefebvre JC, Keefe FJ, Kredich DW, Gil KM. Pain coping and the pain experience in children with juvenile chronic arthritis. Pain1997;73:181–9.[ISI][Medline]
  14. Sherry DD, Bohnsack J, Salmonson K, Wallace CA, Mellins E. Painless juvenile rheumatoid arthritis. J Pediatr Psychol1990;12:241–55.[ISI][Medline]
  15. Ilowite NT, Walco GA, Pochaczevsky R. Assessment of pain in patients with juvenile rheumatoid arthritis: Relation between pain intensity and degree of joint inflammation. Ann Rheum Dis1992;51:343–6.[Abstract]
  16. Thompson KL, Varni JW, Hanson V. Comprehensive assessment of pain in juvenile rheumatoid arthritis: An empirical model. J Pediatr Psychol1987;12:241–55.[ISI][Medline]
  17. Keefe FJ, Brown GK, Wallaston KA, Caldwell DS. Coping with rheumatoid arthritis pain: catastrophizing as a maladaptive strategy. Pain1989;37:51–6.[ISI][Medline]
  18. Newman S, Revenson TA. Coping with rheumatoid arthritis: psychological aspects of rheumatic disease. Balliere's Clin Rheumatol1993;7:259–80.[ISI][Medline]
  19. Fink CW. A proposal for the development of classification criteria for the idiopathic arthritides of childhood. J Rheumatol1995;22:1566–9.[ISI][Medline]
  20. Zigmund AS, Snaith RP. The hospital depression and anxiety scale. Acta Psychiatr Scand1983;67:361–70.[ISI][Medline]
  21. Carr AJ. A patient-centred approach to evaluation and treatment in rheumatoid arthritis: the development of a clinical tool to measure patient-perceived handicap. Br J Rheumatol1996;35:921–32.[ISI][Medline]
  22. Lorig K, Chastain R, Ung E et al. Development and evaluation of a scale to measure perceived self-efficacy in people with arthritis. Arthritis Rheum1989;32:37–44.[ISI][Medline]
  23. Thompson PW, Kirwan JR, Currey HLF. A comparison of the ability of 28 articular indices to detect an induced flare of joint inflammation in rheumatoid arthritis. Br J Rheumatol1988;27:375–80.[ISI][Medline]
  24. Steinbrocker O, Traeger CH, Battman RG. Therapeutic criteria in rheumatoid arthritis. J Am Med Assoc1949;140:659–62.[ISI]
  25. Kirwan JR, Reeback JS. Stanford Health Assessment questionnaire modified to assess disability in British patients with rheumatoid arthritis. Br J Rheumatol1986;25:206–9.[ISI][Medline]
  26. Rosenstiel AK, Keefe FJ. The use of coping strategies in chronic low back pain patients: relationships to patient characteristics and current adjustment. Pain1983;17:33–44.[ISI][Medline]
  27. Sarason I, Levine HM, Basham RB et al. Assessing social support: the social support questionnaire. J Pers Soc Psychol1983;44:127–39.[ISI]
  28. Packham JC, HM. Long term follow up of 246 adults with juvenile idiopathic arthritis:functional outcome. Rheumatology2002;41:1428–35.
  29. Turner RJ, Mclean PD. Physical disability and psychological distress. Rehabil Psychol1989;34:225–42.[ISI]
  30. Timko C, Stovel KW, Moos R, Miller JJ. Adaptation to juvenile rheumatic disease: A controlled evaluation of functional disability with a one-year follow-up. Health Psychol1992;11:91–100.
  31. Gragg RA, Rapoff MA, Danovsky MB et al. Assessing chronic musculoskeletal pain associated with rheumatic disease: further validation of the Pediatric Pain Questionnaire. J Pediatr Psychol1996;21:237–50.[Abstract]
  32. Brainerd CJ. Piaget's theory of intelligence. New Jersey: Prentice Hall,1978.
Submitted 19 June 2002; Accepted 19 July 2002