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Joint Bone Spine
Volume 85, n° 6
pages 733-739 (décembre 2018)
Doi : 10.1016/j.jbspin.2018.01.010
accepted : 30 January 2018
Original Articles

Boundaries between familial Mediterranean fever and juvenile spondyloarthritis: Analysis of three French retrospective cohorts
 

Bilade Cherqaoui a, b, , Linda Rossi-Semerano a, b, Sophie Georgin-Lavialle b, c, Perrine Dusser d, Caroline Galeotti a, b, Maryam Piram a, b, Véronique Hentgen b, e, Isabelle Touitou b, f, Isabelle Koné-Paut a, b
a Pediatric rheumatology, CHU de Bicêtre, AP–HP, 78, rue Gal-Leclerc, 94275 Le Kremlin-Bicêtre, France 
b CeRéMAIA, French reference center for auto-inflammatory diseases and inflammatory amyloidosis, 94270, Le Kremlin Bicêtre, France 
c Internal medicine department, Tenon hospital, AP–HP, 4, rue de la Chine, 75020 Paris, France 
d Pediatric immuno-rheumatology, CHUV Lausanne, 46, rue du Bugnon, 1011 Lausanne, Switzerland 
e Pediatric department, centre hospitalier Le Chesnay, 177, rue de Versailles, 78150 Le Chesnay, France 
f Auto-inflammatory diseases center, CHU de Montpellier, 191 avenue Doyen-Gaston-Giraud, 34090 Montpellier, France 

Corresponding author. Pediatric rheumatology, AP–HP, CHU Bicêtre, 78, rue Gal, Leclerc, 94275 Le Kremlin-Bicêtre, France.Pediatric rheumatology, AP–HP, CHU Bicêtre, 78, rue Gal, Leclerc, 94275 Le Kremlin-Bicêtre, France.
Highlights

Familial Mediterranean fever in children may be associated with typical pattern of juvenile spondyloarthritis.
Children with Familial Mediterranean fever related juvenile spondyloarthritis present a higher rate of inadequate response to colchicine.
Children with Familial Mediterranean fever related juvenile spondyloarthritis should be diagnose earlier and treated as for juvenile spondyloarthritis.
Patients with spondyloarthritis from Mediterranean origins with recurrent extra-articular manifestations should be tested for MEFV gene pathogenic variants.

The full text of this article is available in PDF format.
Abstract
Objectives

Children with Familial Mediterranean fever may suffer from musculoskeletal involvement, somewhat difficult to distinguish from juvenile spondyloarthritis. The association of these two diseases has been scarcely reported in children. Objective of this work was to define the association of familial Mediterranean fever and juvenile spondyloarthritis in France.

Methods

Three cohorts of children with familial Mediterranean fever, juvenile spondyloarthritis, familial Mediterranean fever related juvenile spondyloarthritis, were retrospectively identified in the French reference center of auto-inflammatory diseases. Familial Mediterranean fever was defined according to Tel-Hashomer or Turkish pediatric criteria with at least one exon-10 MEFV -gene mutation. Juvenile spondyloarthritis was defined according to ILAR criteria. Patients with familial Mediterranean fever or juvenile spondyloarthritis were respectively compared to familial Mediterranean fever related juvenile spondyloarthritis patients.

Results

Sixteen children were identified as having familial Mediterranean fever related juvenile spondyloarthritis. The male/female-ratio was 0.6, with median age at spondyloarthritis onset of 7.5years (3–16years). All carried at least one M694V variant in MEFV gene; 16.7% were HLA-B27-carriers. Compared to 83 familial Mediterranean fever patients, familial Mediterranean fever related juvenile spondyloarthritis patients had less frequently fever (P <0.01) and more frequently arthritis (P <0.05), enthesitis (P <0.001), inflammatory back pain (P <0.001), inadequate response to colchicine (P <0.05). Compared to 20 juvenile spondyloarthritis patients, familial Mediterranean fever related juvenile spondyloarthritis patients less often received non-steroidal anti-inflammatory drugs (P <0.01) and anti-tumor necrosis factor drugs (P <0.001).

Conclusions

Familial Mediterranean fever may be associated with typical pattern of juvenile spondyloarthritis. These patients, with less response to colchicine, should be diagnosed earlier and treated as for jSpA.

The full text of this article is available in PDF format.

Keywords : Auto-inflammatory disease, Spondyloarthritis, Juvenile idiopathic arthritis, Colchicine, Anti-TNF drugs, Anti-interleukin 1 drugs




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