Clinical features of children with enthesitis-related juvenile idiopathic arthritis / juvenile spondyloarthritis followed in a French tertiary care pediatric rheumatology centre

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Abstract

Background

Childhood-onset spondyloarthropathies usually start with enthesitis and peripheral arthritis. However, axial disease may develop afterward. Patients are most often classified, following revised (Edmonton 2011) ILAR criteria, as enthesitis-related arthritis, psoriatic arthritis, or unclassified juvenile idiopathic arthritis, particularly in cases of psoriasis in the patient or a first-degree relative. In adults, peripheral spondyloarthritis is classified by ASAS criteria.

Methods

We retrospectively studied patients with childhood-onset spondyloarthropathies followed for more than one year in our referral centre. We did not exclude patients with a personal or familial history of psoriasis.

Results

We included 114 patients followed between January 2008 and December 2015 for a median of 2.5 years (IQR = 2.3). Sixty-nine per-cent of patients fulfilled the revised ILAR classification criteria for enthesitis-related arthritis, and 92% the ASAS criteria for peripheral spondyolarthritis ( p < 0.001). Axial disease and sacroiliitis were rare at disease onset. However, they appeared during follow-up in 63% and 47% of cases respectively, after a median disease duration of 2.6 (IC 95% [2.2–4.4]) and 5.3 years (IC 95% [4.1–7.7]), respectively. Multivariable analysis showed that familial history of spondyloarthritis was associated with the presence of sacroiliitis and active disease at the latest follow-up (OR = 3.61 [1.5–8.7], p < 0.01 and 2.98 [1.2–7.3], p = 0.02, respectively).

Conclusion

Axial involvement developed in most patients within five years. Revised Edmonton criteria were less sensitive than ASAS criteria to classify patients as having childhood-onset spondyloarthropathies. The main risk factor for both sacroiliitis and persistent active disease was a familial history of spondyloarthritis.

Electronic supplementary material

The online version of this article (10.1186/s12969-018-0238-9) contains supplementary material, which is available to authorized users.

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Most cited references 29

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Juvenile idiopathic arthritis.

Angelo Ravelli, Alberto Martini (2007)

Juvenile idiopathic arthritis is a broad term that describes a clinically heterogeneous group of arthritides of unknown cause, which begin before 16 years of age. This term encompasses several disease categories, each of which has distinct methods of presentation, clinical signs, and symptoms, and, in some cases, genetic background. The cause of disease is still poorly understood but seems to be related to both genetic and environmental factors, which result in the heterogeneity of the illness. Although none of the available drugs has a curative potential, prognosis has greatly improved as a result of substantial progresses in disease management. The most important new development has been the introduction of drugs such as anticytokine agents, which constitute a valuable treatment option for patients who are resistant to conventional antirheumatic agents. Further insights into the disease pathogenesis and treatment will be provided by the continuous advances in understanding of the mechanisms connected to the immune response and inflammatory process, and by the development of new drugs that are able to inhibit selectively single molecules or pathways.

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New criteria for inflammatory back pain in patients with chronic back pain: a real patient exercise by experts from the Assessment of SpondyloArthritis international Society (ASAS).

J Sieper, D van der Heijde, R Landewé … (2009)

Inflammatory back pain (IBP) is an important clinical symptom in patients with axial spondyloarthritis (SpA), and relevant for classification and diagnosis. In the present report, a new approach for the development of IBP classification criteria is discussed. Rheumatologists (n = 13) who are experts in SpA took part in a 2-day international workshop to investigate 20 patients with back pain and possible SpA. Each expert documented the presence/absence of clinical parameters typical for IBP, and judged whether IBP was considered present or absent based on the received information. This expert judgement was used as the dependent variable in a logistic regression analysis in order to identify those individual IBP parameters that contributed best to a diagnosis of IBP. The new set of IBP criteria was validated in a separate cohort of patients (n = 648). Five parameters best explained IBP according to the experts. These were: (1) improvement with exercise (odds ratio (OR) 23.1); (2) pain at night (OR 20.4); (3) insidious onset (OR 12.7); (4) age at onset <40 years (OR 9.9); and (5) no improvement with rest (OR 7.7). If at least four out of these five parameters were fulfilled, the criteria had a sensitivity of 77.0% and specificity of 91.7% in the patients participating in the workshop, and 79.6% and 72.4%, respectively, in the validation cohort. This new approach with real patients defines a set of IBP definition criteria using overall expert judgement on IBP as the gold standard. The IBP experts' criteria are robust, easy to apply and have good face validity.

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Long-term outcome in patients with juvenile idiopathic arthritis.

Kirsten Minden, Martina Niewerth, Joachim Listing … (2002)

To describe the long-term outcome of juvenile idiopathic arthritis (JIA). All patients with JIA referred to a pediatric rheumatology center between 1978 and 1988 were identified and invited to undergo an assessment. Patients with JIA from a population-based cohort from East Berlin were included. The outcome assessment considered changes in body function and structure (e.g., mortality, joint abnormalities, disease activity), activities at the individual level (Health Assessment Questionnaire), and participation in society (e.g., mobility, educational and vocational background). Of 260 eligible patients, 215 (83%) were evaluated. Subtypes of JIA at disease onset included oligoarthritis (40%), polyarthritis (14%), systemic arthritis (14%), psoriatic arthritis (1%), enthesitis-related arthritis (15%), and other arthritis (16%). Followup was conducted after a median of 16.5 years. No deaths occurred in this cohort. At followup, approximately half of the patients had active disease and/or changes in body structures to a variable extent. Approximately one-third of patients rated themselves as being functionally limited. Patients demonstrated good social integration: few mobility problems were reported, and the educational achievements of patients were higher and their rate of unemployment was lower compared with the age-matched population. No significant differences in outcome were found between the population-based and the referral-based cohorts. Even though approximately half of the JIA patients had more or less distinctive changes in body function and/or structure after a disease duration of >15 years, fewer than 10% were severely disabled or handicapped. Because JIA often persists into adulthood, long-term followup and care are necessary.

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Author and article information

Contributors

Maxime Goirand:

ORCID: http://orcid.org/0000-0001-5902-446X

33 (0) 1 40 03 41 02 , maxime.goirand@aphp.fr

Journal

Journal ID (nlm-ta): Pediatr Rheumatol Online J

Journal ID (iso-abbrev): Pediatr Rheumatol Online J

Title: Pediatric Rheumatology Online Journal

Publisher: BioMed Central (London )

ISSN (Electronic): 1546-0096

Publication date (Electronic): 2 April 2018

Publication date PMC-release: 2 April 2018

Publication date Collection: 2018

Volume: 16

Electronic Location Identifier: 21

Affiliations

[1 ]ISNI 0000 0001 2175 4109, GRID grid.50550.35, Pediatric Immunology, Hematology, and Rheumatology Unit, Centre de Référence pour les Rhumatismes Inflammatoires et les Maladies Auto-Immunes Systémique Rare de l’Enfant (RAISE) ; Necker-Enfants Malades Hospital, , Assistance Publique Hôpitaux de Paris, ; 149, rue de Sèvres, 75743 Cedex15 Paris, France

[2 ]ISNI 0000 0001 2188 0914, GRID grid.10992.33, Paris Descartes University, ; 12 rue de l’Ecole de Médicine, 75006 Paris, France

[3 ]ISNI 0000 0001 2175 4109, GRID grid.50550.35, Pediatric Radiology Department, Necker-Enfants Malades Hospital, , Assistance Publique Hôpitaux de Paris, ; 149, rue de Sèvres, 75743 Cedex 15 Paris, France

[4 ]ISNI 0000000121496883, GRID grid.11318.3a, UFR SMBH Paris 13, ; 74 rue Marcel Cachin, 93017 Cedex Bobigny, France

[5 ]ISNI 0000 0004 1765 2136, GRID grid.414145.1, Service de Réanimation Néonatale, , Centre Hospitalier Intercommunal de Créteil, ; 40 avenue de Verdun, 94000 Créteil, France

[6 ]GRID grid.462336.6, Imagine Institute, ; 24 boulevard du Montparnasse, 75015 Paris, France

[7 ]ISNI 0000 0001 2175 4109, GRID grid.50550.35, General Pediatrics, Infectious Disease, and Internal Medicine Unit, Robert Debré Hospital, , Assistance Publique Hôpitaux de Paris, ; 48 boulevard Sérurier, 75019 Paris, France

[8 ]ISNI 0000000121866389, GRID grid.7429.8, INSERM UMR 1163, Laboratory of Neurogenetics and Neuroinflammation, ; Paris, France

[9 ]ISNI 0000 0004 1937 0589, GRID grid.413235.2, GOIRAND, CETD et EMASP pédiatrique, , Hôpital Robert Debré, ; 48, Boulevard Serrurier, 75019 Paris, France

Author information

Maxime Goirand http://orcid.org/0000-0001-5902-446X

Article

Publisher ID: 238

DOI: 10.1186/s12969-018-0238-9

PMC ID: 5879929

PubMed ID: 29609643

SO-VID: 67b0d6d2-e8cf-4385-8419-8121ba4213df

License:

Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

History

Date received : 7 November 2017

Date accepted : 13 March 2018

Custom metadata

ScienceOpen disciplines: Pediatrics

Keywords: juvenile spondyloarthritis,enthesitis related arthritis,juvenile idiopathic arthritis,anti-tnf treatment,classification criteria,prognostic factor

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ScienceOpen disciplines: Pediatrics

Keywords: juvenile spondyloarthritis, enthesitis related arthritis, juvenile idiopathic arthritis, anti-tnf treatment, classification criteria, prognostic factor

Clinical features of children with enthesitis-related juvenile idiopathic arthritis / juvenile spondyloarthritis followed in a French tertiary care pediatric rheumatology centre

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Abstract

Background

Methods

Results

Conclusion

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Pediatric Pulse: Advancing Neonatal and Child Health

Most cited references 29

Juvenile idiopathic arthritis.

New criteria for inflammatory back pain in patients with chronic back pain: a real patient exercise by experts from the Assessment of SpondyloArthritis international Society (ASAS).

Long-term outcome in patients with juvenile idiopathic arthritis.

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