{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,9]],"date-time":"2025-10-09T01:10:44Z","timestamp":1759972244822,"version":"build-2065373602"},"reference-count":37,"publisher":"Wiley","issue":"11","license":[{"start":{"date-parts":[[2015,10,28]],"date-time":"2015-10-28T00:00:00Z","timestamp":1445990400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Arthritis &amp; Rheumatology"],"published-print":{"date-parts":[[2015,11]]},"abstract":"<jats:sec><jats:title>Objective<\/jats:title><jats:p>To determine the type and frequency of musculoskeletal symptoms at onset and during followup of cryopyrin\u2010associated periodic syndromes (CAPS).<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>We retrospectively recorded the articular and muscular symptoms of patients with CAPS followed up in French hospitals. Data were presented as frequencies or the median (range), and patient groups were compared using chi\u2010square test, Fisher's exact test, and Mann\u2010Whitney test.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The study included 133 patients (33 children), 20 with familial cold autoinflammatory syndrome, 88 with Muckle\u2010Wells syndrome, 22 with chronic infantile neurologic, cutaneous, articular syndrome, and 3 with unclassified CAPS. The median age was 35 years (range 0\u201378 years) at the time of the study, 1 year (range 0\u201341 years) at symptom onset, and 23 years (range 0\u201358 years) at diagnosis. The disease was sporadic in 17% of the patients. Cutaneous symptoms predominated at onset (77%), followed by articular symptoms (30%). The p.Thr348Met and p.Arg260Trp <jats:italic>NLRP3<\/jats:italic> mutations were significantly associated with the presence and absence of articular symptoms at onset, respectively. During followup, 86% of the patients had musculoskeletal symptoms, 88% had arthralgia, and 58% had arthritis, but only 9% had joint destruction. Tendinopathies occurred in 21.5% of the patients, tender points in 16.5%, and myalgia in 33%. Only 3 patients had typical knee deformities. Radiographs were rarely obtained. Except for bone deformities, osteoarticular symptoms occurred at similar frequencies in the different CAPS phenotypes.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Joint manifestations were frequent in all CAPS phenotypes. Bone deformities were rare. Musculoskeletal manifestations varied within given families but tended to worsen over time.<\/jats:p><\/jats:sec>","DOI":"10.1002\/art.39292","type":"journal-article","created":{"date-parts":[[2015,8,5]],"date-time":"2015-08-05T20:34:33Z","timestamp":1438806873000},"page":"3027-3036","source":"Crossref","is-referenced-by-count":23,"title":["Musculoskeletal Symptoms in Patients With Cryopyrin\u2010Associated Periodic Syndromes: A Large Database Study"],"prefix":"10.1002","volume":"67","author":[{"given":"Laetitia","family":"Houx","sequence":"first","affiliation":[{"name":"Morvan University Hospital Brest France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eric","family":"Hachulla","sequence":"additional","affiliation":[{"name":"Nord\u2010de\u2010France University and Claude\u2010Huriez University Hospital Lille France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Isabelle","family":"Kone\u2010Paut","sequence":"additional","affiliation":[{"name":"Kremlin Bic\u00eatre University Hospital Le Kremlin Bic\u00eatre France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pierre","family":"Quartier","sequence":"additional","affiliation":[{"name":"Necker\u2010Enfants Malades University Hospital and Paris Descartes University Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Isabelle","family":"Touitou","sequence":"additional","affiliation":[{"name":"Arnaud de Villeneuve University Hospital Montpellier France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xavier","family":"Guennoc","sequence":"additional","affiliation":[{"name":"Yves le Foll Hospital Saint\u2010Brieuc France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Gilles","family":"Grateau","sequence":"additional","affiliation":[{"name":"Tenon University Hospital, AP\u2010HP, and Paris VI Pierre et Marie Curie University Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mohamed","family":"Hamidou","sequence":"additional","affiliation":[{"name":"H\u00f4tel Dieu University Hospital Nantes France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"B\u00e9n\u00e9dicte","family":"Neven","sequence":"additional","affiliation":[{"name":"Necker\u2010Enfants Malades University Hospital, AP\u2010HP, and INSERM U768 Laboratory Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jean\u2010Marie","family":"Berthelot","sequence":"additional","affiliation":[{"name":"H\u00f4tel Dieu University Hospital Nantes France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Thierry","family":"Lequerr\u00e9","sequence":"additional","affiliation":[{"name":"Rouen University Hospital Rouen France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pascal","family":"Pillet","sequence":"additional","affiliation":[{"name":"Pellegrin\u2010Enfants University Hospital Bordeaux France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ir\u00e8ne","family":"Lemelle","sequence":"additional","affiliation":[{"name":"University Hospital Nancy France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Michel","family":"Fischbach","sequence":"additional","affiliation":[{"name":"Hautepierre University Hospital Strasbourg France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Agn\u00e8s","family":"Duquesne","sequence":"additional","affiliation":[{"name":"Women\u2010Mothers\u2010Children University Hospital Lyon France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pierre","family":"Le Blay","sequence":"additional","affiliation":[{"name":"Montpellier University Hospital Montpellier France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Claire","family":"Le Jeunne","sequence":"additional","affiliation":[{"name":"Hotel Dieu Hospital Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"J\u00e9rome","family":"Stirnemann","sequence":"additional","affiliation":[{"name":"Jean Verdier University Hospital, AP\u2010HP, Paris XIII University Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christine","family":"Bonnet","sequence":"additional","affiliation":[{"name":"Dupuytren University Hospital Limoges France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Dominique","family":"Gaillard","sequence":"additional","affiliation":[{"name":"Reims University Hospital Reims France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Lilian","family":"Alix","sequence":"additional","affiliation":[{"name":"Rennes University Hospital Rennes France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Renaud","family":"Touraine","sequence":"additional","affiliation":[{"name":"Nord University Hospital Saint\u2010Etienne France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fran\u00e7ois","family":"Garcier","sequence":"additional","affiliation":[{"name":"Saint Chamond  France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christophe","family":"Bedane","sequence":"additional","affiliation":[{"name":"Reims University Hospital Reims France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anne\u2010Laure","family":"Jurquet","sequence":"additional","affiliation":[{"name":"North University Hospital, AP\u2010HM Marseille France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pierre","family":"Duffau","sequence":"additional","affiliation":[{"name":"Saint Andr\u00e9 Hospital, and Bordeaux University Hospital Bordeaux France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Amar","family":"Smail","sequence":"additional","affiliation":[{"name":"North University Hospital Amiens France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Camille","family":"Frances","sequence":"additional","affiliation":[{"name":"Tenon University Hospital, AP\u2010HP Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Martine","family":"Grall\u2010Lerosey","sequence":"additional","affiliation":[{"name":"Pellegrin\u2010Enfants University Hospital Bordeaux France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pascal","family":"Cathebras","sequence":"additional","affiliation":[{"name":"Saint Etienne University Hospital Saint\u2010Etienne France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tu Anh","family":"Tran","sequence":"additional","affiliation":[{"name":"N\u00eemes University Hospital N\u00eemes France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sandrine","family":"Morell\u2010Dubois","sequence":"additional","affiliation":[{"name":"Claude Huriez University Hospital Lille France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Anne","family":"Pagnier","sequence":"additional","affiliation":[{"name":"Toulouse University Hospital Toulouse France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christophe","family":"Richez","sequence":"additional","affiliation":[{"name":"University Hospital Nancy France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Laurence","family":"Cuisset","sequence":"additional","affiliation":[{"name":"Cochin Institute and University Hospital, AP\u2010HP, Paris Descartes University Paris France"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Val\u00e9rie","family":"Devauchelle\u2010Pensec","sequence":"additional","affiliation":[{"name":"La Cavale Blanche University Hospital, Brest and EA2216 Brittany University (UBO) Brest France"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2015,10,28]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1067\/mai.2001.118790"},{"key":"e_1_2_7_3_1","first-page":"235","article-title":"Urticaria, deafness, and amyloidosis: a new heredo\u2010familial syndrome","volume":"31","author":"Muckle T","year":"1962","journal-title":"Q J Med"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(81)80961-4"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00018-003-2296-4"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.10509"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.20633"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2003-07-2531"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1749-6632.2009.05074.x"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.20033"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clinthera.2012.09.009"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.3109\/03009748709102523"},{"key":"e_1_2_7_13_1","first-page":"103","article-title":"A recently recognised chronic inflammatory disease of early onset characterised by the triad of rash, central nervous system involvement and arthropathy","volume":"19","author":"Prieur A.","year":"2001","journal-title":"Clin Exp Rheumatol"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1086\/341357"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0021-9290(00)00056-7"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.10688"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/kel269"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/kel399"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00296-011-2019-3"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.30512"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.21807"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.34409"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa0810787"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1136\/ard.2011.152728"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.25057"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa055137"},{"key":"e_1_2_7_27_1","doi-asserted-by":"publisher","DOI":"10.1136\/ard.2010.138420"},{"key":"e_1_2_7_28_1","doi-asserted-by":"publisher","DOI":"10.2165\/11595040-000000000-00000"},{"key":"e_1_2_7_29_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.27696"},{"key":"e_1_2_7_30_1","doi-asserted-by":"publisher","DOI":"10.1007\/BF00361425"},{"key":"e_1_2_7_31_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11926-011-0165-y"},{"key":"e_1_2_7_32_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00247-006-0358-0"},{"key":"e_1_2_7_33_1","doi-asserted-by":"publisher","DOI":"10.1086\/340786"},{"key":"e_1_2_7_34_1","doi-asserted-by":"publisher","DOI":"10.1097\/00002281-200301000-00011"},{"key":"e_1_2_7_35_1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.112.3.641"},{"key":"e_1_2_7_36_1","first-page":"747","article-title":"Intrafamilial variable phenotypic expression of a CIAS1 mutation: from Muckle\u2010Wells to chronic infantile neurological cutaneous and articular syndrome","volume":"32","author":"Hentgen V","year":"2005","journal-title":"J Rheumatol"},{"key":"e_1_2_7_37_1","doi-asserted-by":"publisher","DOI":"10.1002\/art.27342"},{"key":"e_1_2_7_38_1","first-page":"1","article-title":"Effect of anakinra on arthropathy in CINCA\/NOMID syndrome","volume":"16","author":"Miyamae T","year":"2010","journal-title":"Pediatr Rheumatol Online J"}],"container-title":["Arthritis &amp; Rheumatology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fart.39292","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/acrjournals.onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/art.39292","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,8]],"date-time":"2025-10-08T20:26:13Z","timestamp":1759955173000},"score":1,"resource":{"primary":{"URL":"https:\/\/acrjournals.onlinelibrary.wiley.com\/doi\/10.1002\/art.39292"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,10,28]]},"references-count":37,"journal-issue":{"issue":"11","published-print":{"date-parts":[[2015,11]]}},"alternative-id":["10.1002\/art.39292"],"URL":"https:\/\/doi.org\/10.1002\/art.39292","archive":["Portico"],"relation":{},"ISSN":["2326-5191","2326-5205"],"issn-type":[{"type":"print","value":"2326-5191"},{"type":"electronic","value":"2326-5205"}],"subject":[],"published":{"date-parts":[[2015,10,28]]}}}