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Spectrum of Joint Deformities in Children with Juvenile Idiopathic Arthritis Samia Naz1, Misbah Asif2, Farrah Naz1, Hina Farooq3 and Muhammad Haroon Hamid1

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Spectrum of Joint Deformities in Children with Juvenile Idiopathic Arthritis Samia Naz1, Misbah Asif2, Farrah Naz1, Hina Farooq3 and Muhammad Haroon Hamid1 CLINICAL PRACTICE ARTICLE Spectrum of Joint Deformities in Children with Juvenile Idiopathic Arthritis Samia Naz1, Misbah Asif2, Farrah Naz1, Hina Farooq3 and Muhammad Haroon Hamid1 ABSTRACT Objective: To determine the frequency and types of joint deformities in children with juvenile idiopathic arthritis and their association with clinical parameters and rheumatoid factor. Study Design: Cross-sectional study. Place and Duration of Study: Rheumatology Outpatient Clinic, the Children's Hospital and the Institute of Child Health, Lahore, from September 2014 to February 2015. Methodology: All patients of both genders of less than 16 years of age, who fulfilled the International League of Association for Rheumatology (ILAR) criteria for Juvenile Idiopathic Arthritis (JIA), were enrolled in this study. Their demographic data, duration of disease at the time of presentation, types of JIA, various joint deformities and rheumatoid factor (RF) were documented. Statistical analysis of data was done on SPSS version 16. Chi-square test was applied to determine the association of clinical deformity with age of patients, disease duration at presentation, types of JIA and RF. Results: Out of 70 patients enrolled during the study period, 51.4% were boys with mean age at presentation being 9.44 ±3.89 years (2-7 years) and median duration of disease being 24 months (interquartile range 42 months). Forty patients (57.1%) had joint deformities. Most common joints involved were hand (50%), wrist (50%), and knee (35.7%). The common types of joint deformities were boutonniere deformity (28.6%), ulnar deviation of wrist (28.6%), fixed flexion deformity of wrist (22.9%), and knee (31.4%). The most common type of JIA was polyarthritis RF negative with or without deformity. There was a strong association of deformities with older age of patients at presentation (p=0.036), longer duration of disease at presentation (p=0.028), polyarthritis (RF seronegative / seropositive) (p=0.013), and seropositivity (p=0.04). Conclusion: More than 50% patients with JIA have joint deformities. Joint deformities are more likely to be seen in children with long-standing disease, those with polyarthritis JIA and seropositive patients. Key Words: Juvenile idiopathic arthritis. JIA. Joint deformity. Polyarthritis. Rheumatoid factor (RF). INTRODUCTION spindling of fingers, swan neck deformity, boutonniere Juvenile idiopathic arthritis (JIA) is the leading cause of deformity, Z-deformity of thumb, subluxation of metacarpophalangeal joints, ulnar deviation of wrist, autoimmune arthritis in children and adolescents causing radial deviation of fingers, and flexion/fixed flexion clinical deformities. Epidemiological studies have deformity of wrist. Feet and ankle deformities are lateral reported a burden of 0.07-4.01 per 1000 children.1 The deviation of big toe (hallux valgus), subluxation of true incidence and prevalence in our region is not known. metatarsophalangeal joints and valgus deformity of There are substantial geographic, and ethnic differences ankle. Knee deformities in JIA are valgus and varus are present regarding the frequencies of different types, deformities and flexion/fixed flexion deformity. 1-3 age at onset, and immunological markers. Atlantoaxial subluxtion is the deformity of cervical spine. JIA has different subtypes with varied morbidity. It is a Other orthopedic complications include leg length significant cause of short- and long-term disability in discrepancy and growth delay.5 1,4 children and adolescents. The most serious JIA is a chronic disease causing deformities; and timely complication is the development of joint deformities. diagnosis and prompt multi-disciplinary management is Common deformities of hand and wrist joints include necessary to prevent complications. Various studies have shown different early predictors of poor outcome 1 Department of Paediatric Medicine, Children's Hospital and including female gender, older age at onset, longer The Institute of Child Health, Lahore. duration of disease before referral, early involvement of 2 Department of Physiotherapy, Pakistan Society for the small joints of hands and feet, rapid appearance of Rehabilitation of Disabled, Lahore. erosions, unremitting inflammatory activity, RF 3 Department of Occupational Therapy, Autism Resource Centre, seropositivity, and subcutaneous nodules.6 There is Lahore. paucity of reported literature from Pakistan on this Correspondence: Dr. Samia Naz, Assistant Professor of deforming chronic ailment in children, especially in the Paediatric Medicine, Children's Hospital and The Institute of context of spectrum of deformities and its possible Child Health, Lahore. associations. E-mail: [email protected] The objective of this study was to determine the Received: March 06, 2017; Accepted: March 27, 2018. frequencies and types of joint deformities in juvenile 470 Journal of the College of Physicians and Surgeons Pakistan 2018, Vol. 28 (6): 470-473 Joint deformities in juvenile idiopathic arthritis idiopathic arthritis and their association with clinical and ankles were taken as one, either single or both joints parameters and rheumatoid factor. were involved respectively. Upper limb deformities noted included spindling of fingers, swan neck deformity, METHODOLOGY boutonniere deformity, Z-deformity of thumb, ulnar This cross-sectional analytical study was carried out at deviation of wrist, radial deviation of finger, and Rheumatology Outpatient Clinic, The Children's Hospital flexion/fixed flexion deformity of elbow joint. Lower limb and The Institute of Child Health, Lahore, from deformities noted included flexion/fixed flexion deformity September 2014 to February 2015. Informed consent of knees, valgus deformity of knees and ankles, varus was obtained from all parents or children. All patients deformity of knees, lateral deviation of toes, and outward seen in clinic during the study period, of both genders of deviation of feet. Rheumatoid factor was determined by less than 16 years of age who fulfilled the International indirect haemagglutination method. League of Association for Rheumatology (ILAR) criteria Statistical analysis was performed by statistical package for Juvenile Idiopathic Arthritis (JIA),5 were enrolled in for social sciences (SPSS) version 16.0. Mean and this study. Their demographic data, duration of disease median were determined for quantitative variables. at presentation, and types of arthritis per ILAR criteria Frequencies and percentages were used to describe were recorded in a pretested proforma at their first distribution of age and gender in different groups. clinical visit. The ILAR criteria included the following: Frequency of deformities in various joints of body is 1. Systemic-onset JIA, arthritis in >1 joints with or described as pie chart. Various types of deformities in preceded by fever of at least 2 weeks in duration that is upper and lower limbs are shown as bar charts. Chi- documented to be daily ("quotidian") for at least 3 days square test was performed to determine the relationship and accompanied by >1 of the following: (a) evanescent of demographic data and rheumatoid factor with joint (nonfixed) erythematous rash, (b) generalised lymph deformity. P-value of <0.05 was considered statistically node enlargement, (c) hepatomegaly or splenomegaly significant. Relationship of joint deformities with age, or both, and (d) serositis. duration of disease at presentation, types of arthritis, and RF is described in tabulated form. 2. Oligoarticular JIA, arthritis affecting 1-4 joints during the initial six months of disease. Two subcategories are RESULTS recognised as persistent oligoarthritis-affecting >4 joints throughout the disease course, and extended Out of 70 patients enrolled, 40 (57.1%) were with clinical oligoarthritis-affecting >4 joints after the first 6 months of joint deformities. Among all, 51.4% (n=36) were male disease. and mean age at presentation was 9.44 ±3.89 years (range 2-17 years). Distribution of age at presentation in 3. Rheumatoid factor negative polyarthritis, arthritis various subgroups showed that 8 patients (11.4%) were affecting >5 joints during the initial six months of disease less than 5 years of age, 32 patients (45.7%) were of and a test for RF is negative. 5-10 years, 23 patients (32.9%) were between 11-15 4. Rheumatoid factor positive polyarthritis, arthritis years of age and 7 patients (10%) were more than 15 affecting >5 joints during the initial six month of disease years of age. and 2 or more tests for RF at least 3 months apart during Median duration of disease at presentation was 24 the first 6 months of disease are positive. months with interquartile range 42 months. Distribution 5. Psoriatic arthritis, arthritis and psoriasis, or arthritis of disease duration at presentation showed that 23 and at least 2 of the following: (a) dactylitis, (b) nail pitting patients (32.9%) were presented within 12 months of and onycholysis, (c) psoriasis in a first-degree relative. disease onset, 26 patients (37.1%) between 12-36 6. Enthesitis-related arthritis, arthritis and enthesitis, or months of disease onset, and 21 patients (30.0%) were arthritis or enthesitis with at least two of the following: a. presented after 36 months after disease onset. presence of or a history of sacroiliac joint tenderness Distribution among different subtypes of JIA showed RF- and/or inflammatory lumbosacral pain; b. presence of negative polyarthritis in 43 patients (61.4%), RF-positive HLA-B27 antigen; c. onset of
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