{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,17]],"date-time":"2026-10-17T12:44:48Z","timestamp":1792241088994,"version":"4.4.0"},"reference-count":36,"publisher":"Wiley","issue":"11","license":[{"start":{"date-parts":[[2022,8,24]],"date-time":"2022-08-24T00:00:00Z","timestamp":1661299200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Int J of Rheum Dis"],"published-print":{"date-parts":[[2022,11]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Chronic recurrent multifocal osteomyelitis (CRMO) is a bone inflammatory disorder characterized by osteolytic, usually multiple, symmetric lesions. Diagnosis is one of exclusion, and no standardized therapies are available. Presumed deregulation of the interleukin (IL)\u20101\u03b2 axis, as observed in 2 monogenic autoinflammatory conditions such as Majeed syndrome (LPIN2 mutations) and deficiency of IL\u20101 receptor antagonist (IL1RN mutations) with CRMO\u2010like bone involvement, suggests the blockade of IL\u20101 as potentially useful also in this condition, even if scarce data are available.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation<\/jats:title>\n                    <jats:p>We report the case of a 13\u2010year\u2010old girl affected by a multidrug\u2010resistant and pyoderma gangrenosum\u2010complicated CRMO treated with canakinumab, a human monoclonal antibody targeting IL\u20101\u03b2.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>In this young patient pyoderma gangrenosum and CRMO showed a rapid and satisfactory response to canakinumab, although over time a decreased efficacy in controlling bone disease was observed.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/1756-185x.14425","type":"journal-article","created":{"date-parts":[[2022,8,25]],"date-time":"2022-08-25T02:48:16Z","timestamp":1661395696000},"page":"1333-1338","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Canakinumab treatment in a young girl with refractory chronic recurrent multifocal osteomyelitis associated with pyoderma gangrenosum"],"prefix":"10.1111","volume":"25","author":[{"given":"Sabrina","family":"Acierno","sequence":"first","affiliation":[{"name":"Department of Woman, Child and of General and Specialized Surgery Universit\u00e0 degli Studi della Campania \"Luigi Vanvitelli\"  Naples Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francesca","family":"Angrisani","sequence":"additional","affiliation":[{"name":"Department of Woman, Child and of General and Specialized Surgery Universit\u00e0 degli Studi della Campania \"Luigi Vanvitelli\"  Naples Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Achille","family":"Marino","sequence":"additional","affiliation":[{"name":"Pediatric Rheumatology Unit ASST G. Pini \u2010 CTO  Milan Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9300-6169","authenticated-orcid":false,"given":"Roberto Felice","family":"Caporali","sequence":"additional","affiliation":[{"name":"University of Milan  Milan Italy"},{"name":"Division of Clinical Rheumatology ASST G. Pini \u2010 CTO  Milan Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3260-4226","authenticated-orcid":false,"given":"Rolando","family":"Cimaz","sequence":"additional","affiliation":[{"name":"Pediatric Rheumatology Unit ASST G. Pini \u2010 CTO  Milan Italy"},{"name":"University of Milan  Milan Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9292-7601","authenticated-orcid":false,"given":"Teresa","family":"Giani","sequence":"additional","affiliation":[{"name":"University of Milan  Milan Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,8,24]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1007\/s40272-017-0226-4"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1097\/MOP.0000000000000970"},{"key":"e_1_2_7_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtauto.2021.100095"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1525-1470.1995.tb00122.x"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2133.1995.tb06936.x"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/1346-8138.15417"},{"key":"e_1_2_7_8_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12903-016-0275-z"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neucir.2020.11.007"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1525-1470.1999.00058.x"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.3899\/jrheum.160491"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1007\/s13555-014-0044-3"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1186\/1546-0096-12-30"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0215739"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.1155\/2017\/7694942"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1186\/ar2992"},{"key":"e_1_2_7_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/acr.23462"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1318685111"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1007\/s13555-018-0269-7"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.3390\/biom11030367"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa0807865"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1136\/annrheumdis-2012-201818"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1177\/20587384211006584"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/keq122"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1093\/rheumatology\/keq074"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.3899\/jrheum.160690"},{"key":"e_1_2_7_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.semarthrit.2018.04.003"},{"key":"e_1_2_7_28_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.aller.2016.04.003"},{"issue":"10","key":"e_1_2_7_29_1","article-title":"Canakinumab monotherapy for the treatment of Majeed syndrome. 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