Child: care, health and development

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1 bs_bs_banner Child: care, health and development Original Article doi: /j x Children with juvenile idiopathic arthritis: are health outcomes better for those diagnosed younger?cch_ K. Toupin April,* S. Cavallo and D. Ehrmann Feldman *Department of Epidemiology and Community Medicine, University of Ottawa Centre for Global Health, Institute of Population Health, University of Ottawa, Ottawa, ON Département de médecine sociale et préventive and Institut de recherche en santé publique de l Université de Montréal (IRSPUM), Université de Montréal and Department of Occupational Therapy, Montreal Children s Hospital of the McGill University Health Centre, and École de Réadaptation and Institut de recherche en santé publique de l Université de Montréal (IRSPUM), Université de Montréal, Centre de recherche interdisciplinaire en réadaptation (CRIR), Montreal, QC, Canada Accepted for publication 18 March 2012 Keywords child development, childhood disability, juvenile chronic arthritis, maternal coping, parental adjustment Correspondence Karine Toupin April, Institute of Population Health, University of Ottawa, 1 Stewart Street, Room 201, Ottawa, Ontario, Canada K1N 6N5 ktoupina@uottawa.ca Abstract Background Children living with juvenile idiopathic arthritis (JIA) have swollen, painful and stiff joints, which may have an impact on all spheres of their life, as well as their family. While children diagnosed in their preschool years may be at risk for worse physical outcomes than children diagnosed later in life, it is not clear whether they have worse psychological outcomes and whether these outcomes have an impact on their everyday life. The aim of this study was to examine the association between age at diagnosis and health outcomes in families of children with JIA. Methods Parents of children with JIA (n = 182) who attended rheumatology clinics at the Montreal Children s Hospital and British Columbia Children s Hospital completed measures assessing socio-demographic attributes, their child s health status, health-related quality of life and pain level, as well as their own level of psychological distress and coping behaviours. Regression models explored the association between age at diagnosis and health outcomes while adjusting for sex, age, severity and duration of the disease. Results Parents of children diagnosed with JIA before 5 years of age found their child to have a better health-related quality of life in terms of psychosocial functioning than parents of children diagnosed later [b=-0.91 (-1.63, -0.19)]. Children diagnosed younger were also found to be less limited in schoolwork or activities with friends because of emotional or behavioural problems than children diagnosed later [odds ratio = 0.07 (0.01, 0.42)]. Conclusions Children diagnosed younger seem to show good psychosocial adjustment, which may also be true for their caregivers. Perhaps more attention to psychosocial adjustment should be given to families of children who are diagnosed with the disease at an older age. Introduction Children living with juvenile idiopathic arthritis (JIA) have swollen, painful and stiff joints, which may have an impact on all spheres of their life, as well as their family (Woo & Wedderburn 1998; Schanberg et al. 2003; Petty et al. 2004; Petty & Cassidy 2005). Children s physical and psychological health outcomes may be affected (Flato et al. 2003) while their parents Blackwell Publishing Ltd

2 Outcomes of children diagnosed with JIA at a young age 443 may also feel the impact of the disease or its treatments. The disease may lead to further disability as well as more deformities when children are diagnosed at a young age (Flato et al. 2003). In fact, active disease in the first 5 years of life was shown to be associated with more long-term joint damage, which may lead to greater impairment (Magnani et al. 2009). Furthermore, physical disability in children and adolescents with JIA seems to be related to worse psychological functioning (Ding et al. 2008). While children diagnosed in their preschool years may be at risk for worse physical outcomes than children diagnosed later in life, it is not clear whether they have worse psychological outcomes and whether these outcomes have an impact on their everyday life. Children s health status, health-related quality of life (HRQOL), pain, as well as disease severity are important outcomes to measure in children with JIA when conducting a clinical trial (Giannini et al. 1997). Some of these outcomes incorporate psychological and social dimensions as well as physical ones. Since children diagnosed early with JIA may have different outcomes than children diagnosed later in life, it is essential to understand the relationship between age at diagnosis and health outcomes, especially psychosocial ones. There may also be consequences of the disease on the family (e.g. caregiver psychological distress, parents coping behaviours and families financial hardship) (Timko et al. 1992; Huygen et al. 2000; Bernatsky et al. 2007; Cavallo et al. 2009) and these may differ depending on when the child was diagnosed. The objectives of this study were to examine the association between age at diagnosis with psychological, social and physical health outcomes in families of children with JIA. Methods Study population The study population consisted of all parents of children with JIA aged 2 18 years old who attended the paediatric rheumatology outpatient clinics at the Montreal Children s Hospital and the British Columbia Children s Hospital in Vancouver for at least 6 months. Parents (or primary caregivers living with the child at the time of the study) were asked to participate if they understood either English or French. Ethics approval was obtained from the two institutions and all participating parents signed a consent form. Data collection Parents were asked to answer a series of questionnaires during one of their visits at the clinic or to complete them at home and then return it by mail to the research team. General demographic information and clinical data obtained from the charts included: child s age, sex, type of JIA at diagnosis (e.g. polyarthritis, systemic, oligoarthritis), date of diagnosis, age at diagnosis and disease severity measured by the active joint count (representing the sum of joints with active inflammation as measured by the rheumatologist) (Pediatric Rheumatology Collaborative Study Group 1982). Measures Children s health outcomes Health status was measured using the parent form of the Child Health Questionnaire (CHQ-PF50). The CHQ uses the same structure as the Short Form-36 but was developed specifically for children. It assembles 50 items divided into scales that consider the impact of the child s health on family functioning and scales assessing the child s functioning, pain, behaviours and all spheres of health. Questions of interest to our research team included limitations in schoolwork or activities with friends because of emotional or behavioural problems. The CHQ is a valid and reliable instrument when used for children aged 5 18 years (Landgraf et al. 1998; Nixon et al. 1999; Ruperto et al. 2001; Raat et al. 2002). Health-related quality of life of children with JIA was assessed using the Juvenile Arthritis Quality of Life Questionnaire (JAQQ) (Duffy et al. 1993, 1997, 1995). It includes four domains (gross motor function, fine motor function, psychosocial function and systemic symptoms) and a section assessing pain and global health. Each of the four domains is scored on a Likert scale from 1 to 7, with lower scores indicating less difficulty performing activities. The mean score of a domain is calculated using the five highest scores (which indicate worst quality of life) while the global score of the JAQQ is derived by further calculating the mean of the mean scores. The global score ranges from 1 to 7, with lower scores indicating less difficulty performing activities. Pain in the past week was assessed using a 100 mm visual analogue scale (VAS). The JAQQ has been validated in French and English (Duffy et al. 1997), has excellent sensitivity to change (Duffy et al. 1995) and good construct validity (Duffyet al. 1997). There is also good agreement between parents and children (Toupin April et al. 2006). Parents health outcomes and resources Parent psychological distress was measured with the Symptom Checklist-90-Revised (SCL-90-R), a validated 90-item self-

3 444 K. Toupin April et al. report measure of symptoms experienced by parents during the past week (DeRogatis et al. 1973, 1976; DeRogatis & Cleary 1977a,b; Derogatis 1994). Nine symptoms are assessed: somatization, obsessive compulsive, interpersonal sensitivity, depression, anxiety, hostility, phobic anxiety, paranoid ideation and psychoticism. The sections are summed to create a Global Severity Index, on which T-scores higher than 63 correspond to a clinically important psychological distress (Derogatis 1994). Parental coping with the child s illness was measured with the Child Health Inventory for parents (CHIP) (McCubbin et al. 1983; McCubbin 1987), a 45-item self-report measure by which parents rate their perception of how useful certain coping behaviours are by way of a 4-point Likert scale from not helpful (0) to extremely helpful (3). Coping behaviours are grouped into 3 subscales: (i) maintaining family integration, cooperation and an optimistic definition of the situation (maximum score = 57); (ii) maintaining social support, self-esteem and psychological stability (maximum score = 54); and (iii) understanding the medical situation through communication with other parents and consultation with the medical staff (maximum score = 24). The higher the score the more useful behaviours of a particular type of coping subscale are. The CHIP has good internal consistency and fair concurrent validity (McCubbin et al. 1983; McCubbin 1987). Caregiver perceived family financial situation was assessed by the Economic Hardship Questionnaire (Lempers et al. 1989; Clark-Lempers et al. 1990). This scale consists of 12 items that focus on changes in family s style of living. Internal consistency is excellent but construct validity has not been established. Ten questions are scored on a 4-point Likert scale, with higher scores representing that parents have to cut back on home purchases very often. A score consisting of the sum of these ten questions was computed, leading to a maximum score of 30, indicating high economic hardship. Analysis We classified age into two categories: those diagnosed before 5 years of age and those diagnosed after 5 years of age. This dichotomization has allowed us to assess the impact of diagnosis and disease on children before attending school compared with those diagnosed after having started school. Bivariate analyses between age at diagnosis and psychological, social and physical health outcomes were performed using independent t-test and chi-squared statistics. Outcomes of interest were then explored using multivariate regression models. We used linear regression models for continuous outcomes and logistic regression models for dichotomous outcomes, controlling for the child s sex, age, disease severity and duration. Since the age at diagnosis and the prognosis of JIA are associated with the type of disease at onset (e.g. oligoarthritis patients are younger at diagnosis and have a better prognosis), we also verified if the type of JIA was a confounding variable for each outcome of interest. Results Of the 254 eligible patients, 235 consented to participate in the study: 144 out of the 157 in Montreal, and 91 out of the 97 in Vancouver. In total, 182 parents of children with JIA returned the questionnaires (120 in Montreal and 62 in Vancouver), which corresponds to a response rate of 76.43% for Montreal and 63.92% for Vancouver. The main reason cited for refusing to participate was lack of time to complete the questionnaires. There were no differences between those who participated and those who did not, except for disease severity, which was higher in participants (1.8 vs. 0.6; P = 0.001). Concerning the types of arthritis, 40.4% of children had oligoarthritis, 20.3% polyarthritis, 9% systemic arthritis, 10.1% enthesitis-related arthritis, 11.2% psoriatic arthritis, and 9% had other types of arthritis. Children were prescribed medications in 73.3% of the cases and exercises in 75.6% of the cases. The most commonly used medications were non-steroidal anti-inflammatory drugs (53.9% of participants) followed by disease-modifying anti-rheumatic drugs (41.1%), corticosteroids (8.3%) and TNF-a inhibitors (4.4%). Tables 1 and 2 describe the demographic, psychological and disease-related characteristics of children with JIA and their families. According to bivariate analyses (see Tables 1 & 2), children diagnosed prior to age 5 years were reported to be less limited in schoolwork or activities with friends because of emotional or behavioural problems (9.8% vs. 29.3%, P = 0.02) as per CHQ subtests even though 46% of parents of children diagnosed younger perceived their child s illness as serious whereas only 29.3% of those diagnosed older had this perception (P = 0.04). Also, parents of those diagnosed younger found maintaining social support, a coping behaviour measured by the CHIP, as less useful than those whose children were diagnosed at an older age {mean difference [95% confidence interval (CI)] = 3.91 (0.33, 7.49), P = 0.03}. There were tendencies for other differences as well: those who were diagnosed earlier were perceived as having less pain (as rated on a VAS) [mean difference (95% CI) = 7.20 (-0.01, 14.42), P = 0.05] and families tended to feel more financial hardship [mean difference (95% CI) =-1.81 (-3.64, 0.03), P = 0.05] as per the Economic

4 Outcomes of children diagnosed with JIA at a young age 445 Table 1. Bivariate analyses of children s demographic, health and behavioural outcomes associated with age at disease diagnosis in children with juvenile idiopathic arthritis Children diagnosed before 5 years of age (n = 93) Sex, girl, n (%)* 75 (80.6) 49 (56.3) Age (years), mean SD* Disease duration (years), mean SD* Disease severity (active joint count), mean SD Pain, mean SD Health-related quality of life Total score, mean SD Psychosocial score, mean SD Health status: Limitations in schoolwork or activities with friends because of emotional 6 (9.8) 17 (29.3) difficulties or behavioural problems, parents who perceived some limitations, n (%)* Parents perceived seriousness of the child s illness, parents who perceived some seriousness, n (%)* 29 (46.0) 17 (29.3) Children diagnosed after 5 years of age (n = 87) *Difference statistically significant between the children diagnosed before 5 years old and after: P < Higher scores = higher economic hardship (score 0 30), higher maternal distress, higher disease severity (representing the number of joints with inflammation), higher level of pain (0 100 visual analogue scale) and more difficulty in performing activities (score 1 7). SD, standard deviation. Table 2. Bivariate analyses of parents demographic, health and behavioural outcomes associated with age at disease diagnosis in children with juvenile idiopathic arthritis Children diagnosed before 5 years of age (n = 93) Cultural background, Canadian, n (%) 66 (75) 66 (77.6) Maternal education, higher than high school, n (%) 46 (63.9) 47 (58.0) Economic hardship, mean SD Maternal distress, mean SD Coping behaviours: Maintaining family integration, cooperation and an optimism, mean SD Maintaining social support, self-esteem and psychological stability, mean SD* Understanding the medical situation, mean SD Children diagnosed after 5 years of age (n = 87) *Difference statistically significant between the children diagnosed before 5 years old and after: P < Higher scores = higher economic hardship (score 0 30), higher maternal distress, higher disease severity (representing the number of joints with inflammation), higher level of pain (0 100 visual analogue scale) and more difficulty in performing activities (score 1 7). SD, standard deviation. hardship Questionnaire. HRQOL, physical health as per CHQ items, disease severity and caregiver psychological distress were not associated with age at diagnosis in our sample. When controlling for sex, age, severity and duration of the disease, results of the multivariate analyses indicated that children s psychosocial functioning was significantly associated with age at disease diagnosis (see Table 3). More specifically, parents of children diagnosed younger found their child to have a better HRQOL in terms of psychosocial functioning than parents of children diagnosed later [b=-0.91 (-1.63, -0.19), P = 0.01]. Higher psychosocial functioning was also associated with some of the covariates, namely lower disease severity [b=0.09 (0.04, 0.14), P < 0.001], shorter disease duration [b=0.13 (0.03, 0.22), P = 0.009] as well as older child s age [b =-0.14 (-0.23, 0.05), P = 0.002]. Furthermore, school functioning and activities with friends were also significantly impacted by the child s age at diagnosis. Indeed, children diagnosed with JIA in their preschool years were found by their parents to be less limited in schoolwork or activities with friends because of emotional difficulties or behavioural problems than children diagnosed later [OR = 0.07 (0.01, 0.42), P = 0.004]. Multivariate analyses also showed a non-significant tendency towards diminished psychological distress among parents of children diagnosed younger [b =-7.3 (-14.8, 0.21), P = 0.06].

5 446 K. Toupin April et al. Table 3. Associations between age at diagnosis (and other covariates) and health outcomes and parental coping (multivariate analyses) Outcomes Health-related quality of life psychosocial function, Variables Health children s limitation, OR (95% CI) b (95% CI) Younger age at disease diagnosis 0.07 (0.01, 0.42)* (-1.63, -0.19)* Female sex 0.94 (0.33, 2.70) (-0.75, 0.07) Age (years) 0.80 (0.63, 1.01) (-0.23, -0.05)* Disease duration (years) 1.22 (0.96, 1.54) 0.13 (0.03, 0.22)* Disease severity (active joint count) 1.23 (0.96, 1.59) 0.09 (0.04, 0.14)* *P < OR (95% CI), odds ratio and 95% confidence intervals; b (95% CI), beta coefficients and 95% confidence intervals represent change in the outcome per unit change in the independent variable. These associations remained similar when controlling for the type of arthritis, in addition to the aforementioned covariates. None of the other measured health outcomes were associated with age at diagnosis. In fact, parents appraisal of their child s physical health and pain as well as their own coping behaviours and economic hardship did not vary according to the age at diagnosis. Discussion In this population, children diagnosed in their pre-school years were reported by their parents to be less limited in schoolwork or activities with friends because of psychological difficulties or behavioural problems than those diagnosed later in life. Parents of children diagnosed younger also found their child to have a better HRQOL in terms of psychosocial functioning, which includes interactions with parents, siblings and other children as well as psychological function. These findings seem to corroborate Beales theory of the child s view of chronic illness, which states that younger children see their disease as being less severe and less of a personal disaster than older children (Beales 1983), which may help them to cope with the disease more easily. Other studies in JIA concur with this theory. Huygen et al. have shown that children and adolescents with arthritis seem to cope well with the psychological and social consequences of their condition, which may be explained by an altered frame of reference possibly resulting in a decreased need to compare themselves to their peers (Huygen et al. 2000). Ungerer et al. also suggested that adolescents with JIA may have more difficulty with social relationships than younger children (Ungerer et al. 1988). In our study, parents of children diagnosed earlier than 5 years old felt that their children did not experience worse physical health, HRQOL, pain or adverse psychological outcomes than those diagnosed later, possibly indicating that they too have adjusted to their child s disease. Conversely, parents of children diagnosed at a later age may have more difficulty adapting to the chronic illness since their child had shown the typical level of independence for his/her age up until diagnosis, which will most probably remain the point of comparison for determining their child s state (Andrews et al. 2009). Parents, as primary caregivers, must contend with the ongoing need to manage their child s pain, mobility and activity limitations, and provide regular support and care. Daily treatment, monitoring and dealing with a wide variety of health, social and education issues require much time and attention, which may be especially true for school-aged children. This could explain the observed tendency towards higher psychological distress among parents of children diagnosed at a later time. It is also possible that parents of children diagnosed at a younger age accept the extra responsibility of caring for their child as it is expected for their developmental age, which may make them more resilient than parents of children diagnosed later. By contrast, parents of children diagnosed at a later age must adapt to this new diagnosis for their older child who has been, until the point of diagnosis, independent with respect to everyday activities (e.g. tying their shoelaces, dressing and schoolwork) and may now have difficulty with some of these. Another possible explanation of the worse outcomes in children diagnosed later is that those children may have had the disease for a longer time without being properly diagnosed and treated. Unfortunately, the difference between the time at disease onset or disease symptoms and the time at diagnosis is not known in our sample. Study limitations Parents who accepted to participate in this study may be different from non-participants, leading to a possible selection bias. Participants may have adapted better to the stresses associated with their child s condition, be more motivated, want to learn more

6 Outcomes of children diagnosed with JIA at a young age 447 about their child s state and be more communicative. Parents of children who had the disease younger may have also wanted to share their experience with the researchers, whereas parents of those diagnosed later may remain overwhelmed by their child s diagnosis and be unable to find the time to participate. These characteristics of the responders could reinforce the association between younger age at disease onset and better health outcomes. Also, the cross-sectional nature of our analyses precludes from understanding the relationships between age at disease diagnosis and disease severity longitudinally, which may not reflect the reality of a disease where symptoms vary considerably over time (i.e. consists of flare-ups at unknown intervals). Finally, the date of diagnosis being known but the date of onset of the disease not being known prevents us from establishing the delay in obtaining an accurate diagnosis and a proper treatment, which may have also influenced health outcomes. Conclusion Children who were diagnosed with JIA at a young age were found to have better psychosocial functioning. Their parents may experience a similar positive outcome, where they may feel less psychological distress than parents of children diagnosed at a later age. Children diagnosed younger seem to show good psychosocial adjustment, which may also be true for their caregivers. Perhaps more attention to psychosocial adjustment should be given to children who are diagnosed with the disease at an older age while their parents may require some additional support, given the challenges involved in accepting potential changes in a child s functional status. Key messages Children diagnosed with JIA in their pre-school years were reported to have better psychosocial functioning, while their parents may feel less psychological distress than parents of children diagnosed at a later age. Parents of children diagnosed with JIA in their pre-school years felt that their children did not experience worse physical health, health-related quality of life, pain or adverse psychological outcomes than those diagnosed later, possibly indicating that they have adjusted to their child s disease. More attention to psychosocial adjustment should be given to families with children who are diagnosed with the disease at an older age. Acknowledgements This study was funded by the Canadian Institutes of Health Research, the Sick Kids Foundation, the Canadian Arthritis Network and the Arthritis Society of Canada. Dr Karine Toupin April was supported by graduate training awards from the Canadian Arthritis Network and the Canadian Institutes of Health Research. 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