{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,21]],"date-time":"2026-09-21T03:25:57Z","timestamp":1789961157161,"version":"4.0.1"},"reference-count":20,"publisher":"Bentham Science Publishers Ltd.","issue":"1","license":[{"start":{"date-parts":[[2015,6,26]],"date-time":"2015-06-26T00:00:00Z","timestamp":1435276800000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/3.0\/"}],"content-domain":{"domain":["openrheumatologyjournal.com","benthamopen.com"],"crossmark-restriction":true},"short-container-title":["TORJ"],"published-print":{"date-parts":[[2015,6,26]]},"abstract":"<jats:p>Herein we report successful treatment of an adolescent Caucasian female with severe progressive localized scleroderma (mixed subtype, including generalized morphea and linear scleroderma of the trunk\/limb) using infliximab and leflunomide. The patient demonstrated improvement after the first 9 months of therapy based on her clinical examination, objective measures, and patient and parent global assessments. Infliximab is a potential treatment option for pediatric localized scleroderma patients who have progression of disease or who are unable to tolerate the side effect profile of more standard systemic therapy. Larger longitudinal studies or case series are needed to confirm and further investigate infliximab\u2019s role in localized scleroderma.<\/jats:p>","DOI":"10.2174\/18743129014090100030","type":"journal-article","created":{"date-parts":[[2015,7,6]],"date-time":"2015-07-06T06:27:11Z","timestamp":1436164031000},"page":"30-35","update-policy":"https:\/\/doi.org\/10.2174\/crossmark_policy","source":"Crossref","is-referenced-by-count":31,"title":["A Case Report of Successful Treatment of Recalcitrant Childhood Localized Scleroderma with Infliximab and Leflunomide"],"prefix":"10.2174","volume":"9","author":[{"given":"Ian D","family":"Ferguson","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Peter","family":"Weiser","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathryn S","family":"Torok","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"965","reference":[{"key":"refR1","doi-asserted-by":"crossref","first-page":"381","DOI":"10.1016\/j.pcl.2012.03.011","volume":"59","author":"Torok KS","year":"2012","unstructured":"Torok KS. Pediatric scleroderma systemic or localized forms Pediatr Clin North Am 2012; 59 : 381-405.","journal-title":"Pediatr Clin North Am"},{"key":"refR2","doi-asserted-by":"crossref","first-page":"606","DOI":"10.1097\/01.bor.0000245727.40630.c3","volume":"18","author":"Laxer RM","year":"2006","unstructured":"Laxer RM, Zulian F. Localized scleroderma Curr Opin Rheumatol 2006; 18 : 606-13.","journal-title":"Curr Opin Rheumatol"},{"key":"refR3","doi-asserted-by":"crossref","first-page":"S43","DOI":"10.1002\/art.38444","volume":"66","author":"Wu EY","year":"2014","unstructured":"Wu EY, Li SC, Torok KS , et al. A28: Description of the juvenile localized scleroderma subgroup of the CARRA registry Arthritis Rheumatol 2014; 66 (Suppl 11 ) : S43-44.","journal-title":"Arthritis Rheumatol"},{"key":"refR4","doi-asserted-by":"crossref","first-page":"1044","DOI":"10.1001\/archdermatol.2010.239","volume":"146","author":"Saxton-Daniels S","year":"2010","unstructured":"Saxton-Daniels S, Jacobe HT. An evaluation of long-term outcomes in adults with pediatric-onset morphea Arch Dermatol 2010; 146 : 1044-5.","journal-title":"Arch Dermatol"},{"key":"refR5","doi-asserted-by":"crossref","first-page":"1175","DOI":"10.1002\/acr.21687","volume":"64","author":"Li SC","year":"2012","unstructured":"Li SC, Torok KS, Pope E , et al. Development of consensus treatment plans for juvenile localized scleroderma a roadmap toward comparative effectiveness studies in juvenile localized scleroderma Arthritis Care Res (Hoboken) 2012; 64 : 1175-85.","journal-title":"Arthritis Care Res (Hoboken)"},{"key":"refR6","doi-asserted-by":"crossref","first-page":"91","DOI":"10.1016\/S0022-3476(00)90056-8","volume":"136","author":"Uziel Y","year":"2000","unstructured":"Uziel Y, Feldman BM, Krafchik BR , et al. Methotrexate and corticosteroid therapy for pediatric localized scleroderma J Pediatr 2000; 136 : 91-5.","journal-title":"J Pediatr"},{"key":"refR7","doi-asserted-by":"crossref","first-page":"601","DOI":"10.1001\/archdermatol.2010.120","volume":"146","author":"Diab M","year":"2010","unstructured":"Diab M, Coloe Jr, Magro C , et al. Treatment of recalcitrant generalized morphea with infliximab Arch Dermatol 2010; 146 : 601-4.","journal-title":"Arch Dermatol"},{"key":"refR8","doi-asserted-by":"crossref","first-page":"1433","DOI":"10.1136\/ard.2008.096123","volume":"68","author":"Denton CP","year":"2009","unstructured":"Denton CP, Engelhart M, Tvede N , et al. An open-label pilot study of infliximab therapy in diffuse cutaneous systemic sclerosis Ann Rheum Dis 2009; 68 : 1433-9.","journal-title":"Ann Rheum Dis"},{"key":"refR9","doi-asserted-by":"crossref","first-page":"S31","DOI":"10.1186\/ar1016","volume":"6","author":"De Rycke L","year":"2004","unstructured":"De Rycke L, Baeten D, van den Bosch F , et al. The effect of infliximab on skin lesions in a patient with scleroderma (CREST) Arthritis Res Ther 2004; 6 : S31.","journal-title":"Arthritis Res Ther"},{"key":"refR10","doi-asserted-by":"crossref","first-page":"2819","DOI":"10.3899\/jrheum.081284","volume":"36","author":"Arkachaisri T","year":"2009","unstructured":"Arkachaisri T, Vilaiyuk S, Li S , et al. The localized scleroderma skin severity index and physician global assessment of disease activity a work in progress toward development of localized scleroderma outcome measures J Rheumatol 2009; 36 : 2819-9.","journal-title":"J Rheumatol"},{"key":"refR11","doi-asserted-by":"crossref","first-page":"373","DOI":"10.1093\/rheumatology\/kep361","volume":"49","author":"Arkachaisri T","year":"2010","unstructured":"Arkachaisri T, Vilaiyuk S, Torok KS , et al. Development and initial validation of the localized scleroderma skin damage index and physician global assessment of disease damage a proof-of-concept study Rheumatol (Oxford) 2010; 49 : 373-81.","journal-title":"Rheumatol (Oxford)"},{"key":"refR12","first-page":"S28","volume":"28","author":"Torok KS","year":"2010","unstructured":"Torok KS, Baker NA, Lucas M , et al. Reliability and validity of the delta finger-to-palm (FTP), a new measure of finger range of motion in systemic sclerosis Clin Exp Rheumatol 2010; 28 : S28-36.","journal-title":"Clin Exp Rheumatol"},{"key":"refR13","doi-asserted-by":"crossref","first-page":"904","DOI":"10.1016\/j.jaad.2013.11.037","volume":"70","author":"Das S","year":"2014","unstructured":"Das S, Bernstein I, Jacobe H. Correlates of self-reported quality of life in adults and children with morphea J Am Acad Dermatol 2014; 70 : 904-10.","journal-title":"J Am Acad Dermatol"},{"key":"refR14","doi-asserted-by":"crossref","first-page":"856","DOI":"10.1093\/rheumatology\/kel446","volume":"46","author":"Zulian F","year":"2007","unstructured":"Zulian F, Meneghesso D, Grisan E , et al. A new computerized method for the assessment of skin lesions in localized scleroderma Rheumatol (Oxford) 2007; 46 : 856-60.","journal-title":"Rheumatol (Oxford)"},{"key":"refR15","doi-asserted-by":"crossref","first-page":"S72","DOI":"10.1002\/art.38464","volume":"66","author":"Poff S","year":"2014","unstructured":"Poff S, Li SC, Kelsey C , et al. A48: durometer measures discriminate affected versus normal skin in pediatric localized scleroderma Arthritis Rheumatol 2014; 66 (Suppl 11 ) : S72-3.","journal-title":"Arthritis Rheumatol"},{"key":"refR16","doi-asserted-by":"crossref","first-page":"699","DOI":"10.1002\/art.23564","volume":"59","author":"Merkel PA","year":"2008","unstructured":"Merkel PA, Silliman NP, Denton CP , et al. Validity, reliability, and feasibility of durometer measurements of scleroderma skin disease in a multicenter treatment trial Arthritis Rheumatol 2008; 59 : 699-705.","journal-title":"Arthritis Rheumatol"},{"key":"refR17","doi-asserted-by":"crossref","first-page":"214","DOI":"10.1016\/j.jaad.2013.02.007","volume":"69","author":"Kelsey CE","year":"2013","unstructured":"Kelsey CE, Torok KS. The localized scleroderma cutaneous assessment tool responsiveness to change in a pediatric clinical population J Am Acad Dermatol 2013; 69 : 214-0.","journal-title":"J Am Acad Dermatol"},{"key":"refR18","doi-asserted-by":"crossref","first-page":"548","DOI":"10.1002\/art.20793","volume":"52","author":"Gerloni V","year":"2005","unstructured":"Gerloni V, Pontikaki I, Gattinara M , et al. Efficacy of reApeated intravenous infusions of an anti-tumor necrosis factor alpha monoclonal antibody, infliximab, in persistently active, refractory juvenile idiopathic arthritis results of an open-label prospective study Arthritis Rheumatol 2005; 52 : 548-3.","journal-title":"Arthritis Rheumatol"},{"key":"refR19","doi-asserted-by":"crossref","first-page":"548","DOI":"10.1136\/ard.2006.058248","volume":"66","author":"Tynjala P","year":"2007","unstructured":"Tynjala P, Lindahl P, Honkanen V , et al. Infliximab and etanercept in the treatment of chronic uveitis associated with refractory juvenile idiopathic arthritis Ann Rheumatol Dis 2007; 66 : 548-0.","journal-title":"Ann Rheumatol Dis"},{"key":"refR20","doi-asserted-by":"crossref","first-page":"718","DOI":"10.1136\/ard.2009.100354","volume":"69","author":"Ruperto N","year":"2010","unstructured":"Ruperto N, Lovell DJ, Cuttica R , et al. Long-term efficacy and safety of infliximab plus methotrexate for the treatment of polyarticular-course juvenile rheumatoid arthritis findings from an open-label treatment extension Ann Rheumatol Dis 2010; 69 : 718-22.","journal-title":"Ann Rheumatol Dis"}],"container-title":["The Open Rheumatology Journal"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/openrheumatologyjournal.com\/contents\/volumes\/V9\/TORJ-9-30\/TORJ-9-30.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/openrheumatologyjournal.com\/contents\/volumes\/V9\/TORJ-9-30\/TORJ-9-30.xml","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/openrheumatologyjournal.com\/contents\/volumes\/V9\/TORJ-9-30\/TORJ-9-30.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,6,9]],"date-time":"2024-06-09T21:29:44Z","timestamp":1717968584000},"score":1,"resource":{"primary":{"URL":"https:\/\/openrheumatologyjournal.com\/VOLUME\/9\/PAGE\/30\/"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,6,26]]},"references-count":20,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2015,6,26]]}},"alternative-id":["TORJ-9-30"],"URL":"https:\/\/doi.org\/10.2174\/18743129014090100030","relation":{},"ISSN":["1874-3129"],"issn-type":[{"value":"1874-3129","type":"print"}],"subject":[],"published":{"date-parts":[[2015,6,26]]},"assertion":[{"value":"Peer Reviewed","order":0,"name":"review_status","label":"Review Status","group":{"name":"peer_review_details","label":"Peer Review Details"}},{"value":"Single blind","order":1,"name":"review_process","label":"Review Process","group":{"name":"peer_review_details","label":"Peer Review Details"}},{"value":"Checked with iThenticate","order":0,"name":"screening_status","label":"Screening Status","group":{"name":"plagiarism_screening","label":"Plagiarism Screening"}},{"value":"2014-12-9","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2015-2-17","order":1,"name":"revised","label":"Revised","group":{"name":"publication_history","label":"Publication History"}},{"value":"2015-2-27","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2015-6-26","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}