The Canadian English and French versions of the Juvenile Arthritis Multidimensional Assessment Report (JAMAR)

The Juvenile Arthritis Multidimensional Assessment Report (JAMAR) is a new parent/patient reported outcome measure that enables a thorough assessment of the disease status in children with juvenile idiopathic arthritis (JIA). We report the results of the cross-cultural adaptation and validation of the parent and patient versions of the JAMAR in the Canadian varieties of English and French. The reading comprehension of the questionnaires were tested in a probe sample of ten parents and ten JIA patients for Canadian English and other ten parents and ten JIA patients for Canadian French. Each participating centre was asked to collect demographic, clinical data and the JAMAR in 100 consecutive JIA patients or all consecutive patients seen in a 6-month period and to administer the JAMAR to 100 healthy children and their parents. The statistical validation phase explored descriptive statistics and the psychometric issues of the JAMAR: the 3 Likert assumptions, floor/ceiling effects, internal consistency, Cronbach’s alpha, interscale correlations, test–retest reliability and construct validity (convergent and discriminant validity). A total of 208 JIA patients (2.9% systemic, 41.8% oligoarticular, 27.9% RF negative polyarthritis, 27.4% other categories) and 152 healthy children, were enrolled at two paediatric rheumatology centres. The JAMAR components discriminated well healthy subjects from JIA patients. Notably, there was no significant difference between the healthy subjects and their affected peers in the psychosocial quality of life variable. All JAMAR components revealed good psychometric performances. In conclusion, the Canadian English and French versions of the JAMAR are valid tools for the assessment of children with JIA and are suitable for use both in routine clinical practice and clinical research.


Introduction
The aim of the present study was to cross-culturally adapt and validate the Canadian English and French parent, child/ adult version of the Juvenile Arthritis Multidimensional Assessment Report (JAMAR) [1] in patients with juvenile idiopathic arthritis (JIA). The JAMAR assesses the most relevant parent/patient reported outcomes in JIA, including overall well-being, functional status, health related quality of life (HRQoL), pain, morning stiffness, disease activity/ status/course, articular and extra-articular involvement, drug-related side effects/compliance and satisfaction with illness outcome.
This project was part of a larger multinational study conducted by the Paediatric Rheumatology International Trials Organisation (PRINTO) [2] aimed to evaluate the Epidemiology, Outcome and Treatment of Childhood Arthritis (EPOCA) in different geographic areas [3].
We report herein the results of the cross-cultural adaptation and validation of the parent and patient versions of the JAMAR in the Canadian varieties of English and French.

Materials and methods
The methodology employed has been described in detail in the introductory paper of the supplement [4]. In brief, it was a cross-sectional study of JIA children, classified according to the ILAR criteria [5,6] and enrolled from February 2012 to June 2016. Children were recruited after Ethics Committee approval and consent from at least one parent.

The JAMAR
The JAMAR (1) includes the following 15 sections: 1. Assessment of physical function (PF) using 15-items in which the ability of the child to perform each task is scored as follows: 0 = without difficulty, 1 = with some difficulty, 2 = with much difficulty, 3 = unable to do and not applicable if it was not possible to answer the question or the patient was unable to perform the task due to their young age or to reasons other than JIA. The total PF score ranges from 0 to 45 and has 3 components: PF-lower limbs (PF-LL); PF-hand and wrist (PF-HW) and PF-upper segment (PF-US) each scoring from 0 to 15 [7]. Higher scores indicating higher degree of disability [8][9][10] 12. Report of school/university/work problems caused by the disease (list of items). 13. Assessment of HRQoL, through the Physical Health (PhH), and Psychosocial Health (PsH) subscales (five items each) and a total score. The four-point Likert response, referring to the prior month, are 'never' (score = 0), 'sometimes' (score = 1), 'most of the time' (score = 2) and 'all the time' (score = 3). A 'not assessable' column was included in the parent version of the questionnaire to designate questions that cannot be answered because of developmental immaturity. The total HRQoL score ranges from 0 to 30, with higher scores indicating worse HRQoL. A separate score for PhH and PsH (range 0-15) can be calculated [12][13][14]. 14. Rating of the patient's overall well-being on a 21-numbered circle VAS. 15. A question about satisfaction with the outcome of the illness (yes/no) [15].
The JAMAR is available in three versions, one for parent proxy-report (child's age 2-18), one for child self-report, with the suggested age range of 7-18 years, and one for adults.

Cross-cultural adaptation and validation
The process of cross-cultural adaptation was conducted according to international guidelines with 2-3 forward and backward translations. In those countries for which the translation of JAMAR had been already cross-cultural adapted in a similar language (i.e. Spanish in South American countries), only the probe technique was performed. Reading comprehension and understanding of the translated questionnaires were tested in a probe sample of ten parents and ten JIA patients for Canadian English and other ten parents and ten JIA patients for Canadian French.
Each participating centre was asked to collect demographic, clinical data and the JAMAR in 100 consecutive JIA patients or all consecutive patients seen in a 6-month period and to administer the JAMAR to 100 healthy children and their parents.
The statistical validation phase explored the descriptive statistics and the psychometric issues [16]. In particular, we evaluated the following validity components: the first Likert assumption [mean and standard deviation (SD) equivalence]; the second Likert assumption or equal items-scale correlations (Pearson r: all items within a scale should contribute equally to the total score); third Likert assumption (item internal consistency or linearity for which each item of a scale should be linearly related to the total score that is 90% of the items should have Pearson r ≥ 0.4); floor/ceiling effects (frequency of items at lower and higher extremes of the scales, respectively); internal consistency, measured by the Cronbach's alpha, interscale correlation (the correlation between two scales should be lower than their reliability coefficients, as measured by Cronbach's alpha); test-retest reliability or intra-class correlation coefficient (reproducibility of the JAMAR repeated after 1 or 2 weeks); and construct validity in its two components: the convergent or external validity which examines the correlation of the JAMAR subscales with the six JIA core set variables, with the addition of the parent assessment of disease activity and pain by the Spearman's correlation coefficients (r) [17] and the discriminant validity, which assesses whether the JAMAR discriminates between the different JIA categories and healthy children [18].
Quantitative data were reported as medians with 1st and 3rd quartiles and categorical data as absolute frequencies and percentages.
The complete Canadian English and French translations of JAMAR (parent and patient versions) are available upon request to PRINTO.

Cross cultural adaptation
The Canadian English JAMAR was fully cross-culturally adapted from the original standard English version of the questionnaire with no forward and backward translation.
The Canadian French JAMAR was fully cross-culturally adapted from the Swiss French version performed by the PRINTO centre in Switzerland with no forward and backward translation.
With regard to the Canadian English JAMAR, all 123 lines of the parent version of the JAMAR were understood by at least 80% of the 10 parents tested (median 100%; range 90-100%) and all the 120 lines of the patient version of the JAMAR were understood by at least 80% of the children (median 100%; range 100-100%). The parent and child versions of the Canadian English were unmodified after the probe technique.
With regard to the Canadian French JAMAR, of the 123 lines in the parent version of the JAMAR, 122 (99.2%) were understood by at least 80% of the 10 parents tested (median 100%; range 70-100%). All the 120 lines of the patient version of the JAMAR were understood by at least 80% of the children (median 100%; range 80-100%). Line 54 in the parent version of the JAMAR was modified according to parent's suggestions; the child versions of the Canadian English was unmodified after the probe technique.

Demographic and clinical characteristics of the subjects
A total of 208 JIA patients and 152 healthy children (total of 360 subjects), were enrolled at two paediatric rheumatology centres in Canada.
A total of 354/360 (98.3%) subjects had the parent version of the JAMAR completed by a parent (206 from parents of JIA patients and 148 from parents of healthy children). The JAMAR was completed by 296/354 (83.6%) mothers and 58/354 (16.4%) fathers. The child version of the JAMAR was completed by 307/360 (85.3%) children age 6.8 or older.

Discriminant validity
The JAMAR results are presented in Table 1, including the scores [median (1st-3rd quartile)] obtained for the PF, the PhH, the PsH subscales and total score of the HRQoL scales. The JAMAR components discriminated well between healthy subjects and JIA patients.
In summary, the JAMAR revealed that JIA patients had a greater level of disability and pain, as well as a lower HRQoL than their healthy peers. However, there was no significant difference between healthy subjects and their affected peers in HRQoL PsH variable.

Psychometric issues
The main psychometric properties of both parent and child versions of the JAMAR are reported in Table 2. The following results section refers mainly to the parent's version findings, unless otherwise specified.

Descriptive statistics (first Likert assumption)
For all JAMAR items, the median number of missing responses was 3.9% (3.9-4.4%). The response pattern for both PF and HRQoL was positively skewed toward normal functional ability and normal HRQoL. All response choices were used for the different HRQoL items except for item 1, whereas a reduced number of response choices was used for all the PF items with the exception of items 1, 3, 4, and 13.

Equal items-scale correlations (second Likert assumption)
Pearson items-scale correlations corrected for overlap were roughly equivalent for items within a scale for 93% of the

Items internal consistency (third Likert assumption)
Pearson items-scale correlations were ≥ 0.4 for 87% of items of the PF (except for items 11 and 15) and 90% of items of the HRQoL (except for item 1).

Interscale correlation
The Pearson correlation of each item of the PF and the HRQoL with all items included in the remaining scales of the questionnaires was lower than the Cronbach's alpha, with the exception of PF item 14.

Test-retest reliability
Reliability was assessed in 18 JIA patients, by re-administering both versions (parent and child) of the JAMAR after a median of 5.5 days (4-7 days). The intraclass correlation coefficients (ICC) for the PF total score showed an almost perfect reproducibility (ICC 0.97). The ICC for the HRQoL PhH score and for the HRQoL PsH score showed an almost perfect reproducibility (ICC 1.0 for both).

Convergent validity
The Spearman correlation of the PF total score with the JIA core set of outcome variables ranged from 0.4 to 0.6 (median 0.5). The PF total score best correlation was observed with the parent's assessment of pain (r = 0.7, p < 0.001). For the HRQoL, the median correlation of the PhH with the JIA core set of outcome variables ranged from 0.3 to 0.7 (median 0.5), whereas for the PsH ranged from 0.2 to 0.6 (median 0.4). The PhH showed the best correlation with the parent's assessment of pain (r = 0.8, p < 0.001) and the PsH with the parent global assessment of well-being (r = 0.6, p < 0.001).
The median correlations between the pain VAS, the wellbeing VAS, and the disease activity VAS and the physician-

Discussion
In this study, the Canadian English JAMAR was fully crossculturally adapted from the original standard English version with no forward and backward translation and the Canadian French JAMAR was fully cross-culturally adapted from the Swiss French version performed by the PRINTO centre in Switzerland with no forward and backward translation.
According to the results of the validation analysis, the Canadian parent and patient versions of the JAMAR possess satisfactory psychometric properties. The disease-specific components of the questionnaire discriminated well between patients with JIA and healthy controls. Notably, there was no significant difference between the healthy subjects and their affected peers in the psychosocial quality of life variable. This finding indicates that children with JIA adapt well to the consequences of JIA.
Psychometric performances were good for all domains of the JAMAR with few exceptions: PF items 11 and 15 ("stretch out arms" and "bite a sandwich or an apple") and HRQoL item 1 ("Difficulty of taking care of him/herself") showed a lower items internal consistency. However, the overall internal consistency was at least acceptable for all the domains.
In the external validity evaluation, the Spearman's correlations of the PF and HRQoL scores with JIA core set parameters range from weak to moderate.
When we analyze the data of the JAMAR questionnaire separately, dividing the French-speaking population from the Engligh one, the results were substantially overlapping (data not shown).
The statistical performances of the child version of the JAMAR are very similar to those obtained by the parent version, which suggests that children are reliable reporters of their disease and health status.
The JAMAR is aimed to evaluate the side effects of medications and school attendance, which are other dimensions of daily life that were not previously considered by other HRQoL tools. This may provide useful information for intervention and follow-up in health care.
In conclusion, the Canadian English and French versions of the JAMAR were found to have satisfactory psychometric properties and represent, thus, reliable and valid tools for the multidimensional assessment of children with JIA.