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      Takayasu arteritis with an initial presentation of chronic monoarthritis mimicking oligoarticular juvenile idiopathic arthritis

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          Abstract

          Patients with Takayasu arteritis (TA) generally present with non-specific symptoms that, if unrecognized and untreated, may develop vessel stenosis and/or aneurysm. There is limited data regarding chronic monoarthritis as the initial presentation in children with TA. We report a 6-yearold girl diagnosed and treated as oligoarticular juvenile idiopathic arthritis (JIA). She later developed stroke with malignant hypertension and was definitively diagnosed with TA. She additionally developed proteinuria secondary to focal segmental glomerulosclerosis. This is the report of a patient with chronic monoarthritis mimicking oligoarticular JIA which chronic monoarthritis was the presentation of TA. Since JIA is a diagnosis of exclusion, any atypical features of oligoarticular JIA should illuminate the possibility of an alternative diagnosis. Our literature review focused on musculoskeletal presentations of children with TA.

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          Takayasu arteritis in childhood: retrospective experience from a tertiary referral centre in the United Kingdom

          Despina Eleftheriou, Giulia Varnier, Pavla Dolezalova … (2015)
          Introduction Takayasu arteritis (TA) is an idiopathic large-vessel vasculitis affecting the aorta and its major branches. Although the disease rarely affects children, it does occur, even in infants. The objective of this study was to evaluate the clinical features, disease activity, treatment and outcome of childhood TA in a tertiary UK centre. Methods We analysed a retrospective case series of children fulfilling the TA classification criteria of the European League against Rheumatism, the Paediatric Rheumatology European Society and the Paediatric Rheumatology International Trials Organisation. Data regarding demographics, clinical features, treatments and outcomes were recorded. Descriptive statistics are expressed as median and range. Fisher’s exact test was used for group comparisons. The Paediatric Vasculitis Activity Score (PVAS), Paediatric Vasculitis Damage Index (PVDI), Disease Extent Index-Takayasu (DEI.Tak) and Indian Takayasu Arteritis Activity Score (ITAS2010) were calculated retrospectively. Results A total of 11 children (64% female) with age at diagnosis of 11.8 (1.3 to 17) years were identified over a 23-year period. The median time to diagnosis was 17 (0 to 132) months. The most common clinical features at presentation were arterial hypertension (72.7%), systemic features (36%) and cardiovascular (45%), neurological (36%), pulmonary (27%), skin (9%), renal (9%) and gastrointestinal (9%) involvement. At presentation, PVAS was 5/63 (1 to 13); DEI.Tak was 7/81 (2 to 12) and ITAS2010 was 9/57 (6 to 20). Treatment included corticosteroids (81.8%), combined with methotrexate in most cases (72.7%). Cyclophosphamide (36.4%) and biologic agents (45.5%) were reserved for severe and/or refractory cases. PVDI at latest follow-up was 5.5/72 (3 to 15). Mortality was 27%. Young age at disease onset (<5 years old) and permanent PVDI scores ≥3 were significantly associated with mortality risk (P = 0.024). Conclusion TA is a rare and potentially life-threatening large-vessel vasculitis. Improved awareness of TA is essential to secure a timely diagnosis. Although the evidence base for the treatment of TA in children is weak, we found that it is essential to treat it aggressively because our data emphasise that the mortality and morbidity in the paediatric population remains high. Electronic supplementary material The online version of this article (doi:10.1186/s13075-015-0545-1) contains supplementary material, which is available to authorized users.
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            Childhood Takayasu arteritis: disease course and response to therapy

            Florence Aeschlimann, Simon Eng, Shehla Sheikh … (2017)
            Background Takayasu arteritis (TAK) is a large vessel vasculitis that rarely affects children. Data on childhood TAK are scarce. The aim of this study was to analyze the presenting features, course and outcome of children with TAK, compare efficacy of treatment regimens and identify high-risk factors for adverse outcome. Methods A single-center cohort study of consecutive children fulfilling the EULAR/PRINTO/PReS criteria for childhood TAK between 1986 and 2015 was performed. Clinical phenotypes, laboratory markers, imaging features, disease course and treatment were documented. Disease activity was assessed using the Pediatric Vasculitis Disease Activity Score at each visit. Outcome: disease flare defined as new symptoms and/or increased inflammatory markers necessitating therapy escalation and/or new angiographic lesions, or death. Analysis: logistic regression tested relevant variables for flare. Kaplan-Meier analyses compared treatment regimens. Results Twenty-seven children were included; 74% were female, median age at diagnosis was 12.4 years. Twenty-two (81%) children presented with active disease at diagnosis. Treatment regimens included corticosteroids alone (15%), corticosteroids plus methotrexate (37%), cyclophosphamide (19%), or a biologic agent (11%). Adverse outcomes were documented in 14/27 (52%) children: two (7%) died within 6 months of diagnosis, and 13 (48%) experienced disease flares. The 2-year flare-free survival was 80% with biologic treatments compared to 43% in non-biologic therapies (p = 0.03); at last follow-up, biologic therapies resulted in significantly higher rates of inactive disease (p = 0.02). No additional outcome predictor was identified. Conclusions Childhood TAK carries a high disease burden; half of the children experienced flares and 7% died. Biologic therapies were associated with better control of disease activity.
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              Takayasu arteritis in children and young indians.

              S. Jain, N. Sharma, S. Singh … (2000)
              Twenty four patients of Takayasu arteritis (TA) aged less than 18 years were studied over a period of 20 years (1978-98). There were 4 males and 20 females with a male:female ratio of 1:5. The mean age of presentation was 14 years and the disease had a mean onset of time 4+/-1.5 months prior to admission to the hospital. Hypertension was the commonest mode of presentation seen in 83% of patients. 16% patients had congestive heart failure. Left ventricular hypertrophy was present in 54% patients. Angiographic findings showed that abdominal aorta was the commonest segment of aorta that was involved (71% cases). Renal artery was involved in 75% cases. Treatment modalities included antihypertensive drugs in 19 patients, antitubercular drugs in 7 patients and steroids in 7 patients. Renal angioplasty was performed in 2 patients with excellent results. On follow up 2 patients died. The causes of mortality were renal failure and heart failure in one patient each. The clinical profile of young patients with TA is similar to that of adults with this disease.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Pediatr Rep
                Journal ID (publisher-id): PR
                Title: Pediatric Reports
                Publisher: PAGEPress Scientific Publications, Pavia, Italy
                ISSN (Print): 2036-749X
                ISSN (Electronic): 2036-7503
                Publication date (Electronic): 09 July 2018
                Publication date Collection: 24 May 2018
                Volume: 10
                Issue: 2
                Electronic Location Identifier: 7648
                Affiliations
                [1 ]Division of Rheumatology
                [2 ]Division of Neurology
                [3 ]Division of Nephrology, Department of Pediatrics, Faculty of Medicine, Siriraj Hospital, Mahidol University , Bangkok, Thailand
                Author notes
                Division of Rheumatology, Department of Pediatrics, Faculty of Medicine, Siriraj Hospital, Mahidol University, 2 Wanglang Road, Bangkoknoi, Bangkok 10700, Thailand. +66.2.419.5655 - +66.2.419.5960. sirirat.chv@ 123456mahidol.ac.th

                Contributions: MS and SC drafted the manuscript. SV, NP, and SC contributed to the writing and reviewing of the manuscript. All authors read and approved the final manuscript.

                Conflict of interest: the authors declare no potential conflict of interest.

                Article
                DOI: 10.4081/pr.2018.7648
                PMC ID: 6050474
                SO-VID: e57b2ffc-ea3e-42b3-a204-f2b4e0d93d81
                Copyright © ©Copyright M. Sukharomana et al., 2018
                License:

                This article is distributed under the terms of the Creative Commons Attribution Noncommercial License ( by-nc 4.0) which permits any noncommercial use, distribution, and reproduction in any medium, provided the original author(s) and source are credited.

                History
                Date received : 21 February 2018
                Date accepted : 24 June 2018
                Page count
                Figures: 2, Tables: 1, Equations: 0, References: 17, Pages: 4
                Funding
                Funding: none.
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Pediatrics
                Keywords: takayasu arteritis,oligoarticular juvenile idiopathic arthritis,focal segmental glomerulosclerosis,hypertension,stroke
                Data availability:
                ScienceOpen disciplines: Pediatrics
                Keywords: takayasu arteritis, oligoarticular juvenile idiopathic arthritis, focal segmental glomerulosclerosis, hypertension, stroke

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