{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,1]],"date-time":"2026-05-01T23:50:13Z","timestamp":1777679413627,"version":"3.51.4"},"reference-count":8,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2026,1,9]],"date-time":"2026-01-09T00:00:00Z","timestamp":1767916800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,3,31]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Dupilumab, an interleukin (IL)-4 receptor alpha antagonist, is widely used in the treatment of atopic dermatitis and has shown potential benefit in certain fibrosing skin conditions. While blockade of IL-4 and IL-13 is generally considered to inhibit fibrosis, emerging reports of localized sclerosing dermatoses paradoxically arising during dupilumab therapy suggest a more complex immunological effect. We report a 37-year-old woman with the first case of systemic sclerosis developing in a patient with atopic dermatitis treated with dupilumab. After 2 years of treatment, she presented with new-onset dyspnoea, Raynaud phenomenon and digital puffiness. Serological tests revealed positive antinuclear and anticentromere antibodies; thoracic computed tomography showed interstitial lung disease. The modified Rodnan skin score was 4, and a skin biopsy from the fingertip demonstrated compact hyperkeratosis, irregular acanthosis and fibrosis extending to the superficial dermis. The patient met the American College of Rheumatology\/European Alliance of Associations for Rheumatology 2013 classification criteria for systemic sclerosis, with a total score of 10. Dupilumab was discontinued, and systemic treatments targeting vascular symptoms and pulmonary involvement were initiated, resulting in clinical improvement. This case highlights a significant potential paradoxical reaction associated with cytokine-targeted biologic therapy. It underscores the importance of monitoring for fibrotic and systemic symptoms even when using agents presumed to exert antifibrotic effects.<\/jats:p>","DOI":"10.1093\/skinhd\/vzaf100","type":"journal-article","created":{"date-parts":[[2025,11,5]],"date-time":"2025-11-05T13:45:49Z","timestamp":1762350349000},"page":"160-163","source":"Crossref","is-referenced-by-count":1,"title":["Unexpected systemic sclerosis in a patient with atopic dermatitis receiving dupilumab: a novel case report"],"prefix":"10.1093","volume":"6","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6368-4804","authenticated-orcid":false,"given":"Ela","family":"Gazal","sequence":"first","affiliation":[{"name":"Department of Dermatology, Mersin University Hospital , Mersin ,","place":["T\u00fcrkiye"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Saffet Burak","family":"Ba\u015fak","sequence":"additional","affiliation":[{"name":"Department of Dermatology, Mersin University Hospital , Mersin ,","place":["T\u00fcrkiye"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yasemin","family":"Yuyucu Karabulut","sequence":"additional","affiliation":[{"name":"Department of Pathology, Mersin University Hospital , Mersin ,","place":["T\u00fcrkiye"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"\u00dcmit","family":"T\u00fcrsen","sequence":"additional","affiliation":[{"name":"Department of Dermatology, Mersin University Hospital , Mersin ,","place":["T\u00fcrkiye"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2026,1,9]]},"reference":[{"key":"2026033107425852900_vzaf100-B1","doi-asserted-by":"crossref","first-page":"154799","DOI":"10.1016\/j.cyto.2019.154799","article-title":"Interleukin-4 and interleukin-13 as possible therapeutic targets in systemic sclerosis","volume":"125","author":"Gasparini","year":"2020","journal-title":"Cytokine"},{"key":"2026033107425852900_vzaf100-B2","doi-asserted-by":"crossref","first-page":"e161","DOI":"10.1111\/jdv.16097","article-title":"Keloid lesions show increased IL-4\/IL-13 signaling and respond to Th2-targeting dupilumab therapy","volume":"34","author":"Diaz","year":"2020","journal-title":"J Eur Acad Dermatol Venereol"},{"key":"2026033107425852900_vzaf100-B3","doi-asserted-by":"crossref","first-page":"501193","DOI":"10.1016\/j.abd.2025.501193","article-title":"Paradoxical morphea-like reaction after initiation of dupilumab for nodular prurigo","volume":"100","author":"Gonz\u00e1lez Fern\u00e1ndez","year":"2025","journal-title":"An Bras Dermatol"},{"key":"2026033107425852900_vzaf100-B4","doi-asserted-by":"crossref","first-page":"540","DOI":"10.1111\/pde.15200","article-title":"Morphea after initiation of dupilumab in two pediatric atopic dermatitis patients","volume":"40","author":"Wang","year":"2023","journal-title":"Pediatr Dermatol"},{"key":"2026033107425852900_vzaf100-B5","doi-asserted-by":"crossref","first-page":"7","DOI":"10.1016\/j.jdcr.2021.08.018","article-title":"A case of paradoxical fibrosis and development of morphea in a patient on dupilumab","volume":"17","author":"Kassamali","year":"2021","journal-title":"JAAD Case Rep"},{"key":"2026033107425852900_vzaf100-B6","doi-asserted-by":"crossref","first-page":"262","DOI":"10.1007\/s12016-021-08889-8","article-title":"Current concepts on the pathogenesis of systemic sclerosis","volume":"64","author":"Truchetet","year":"2023","journal-title":"Clin Rev Allergy Immunol"},{"key":"2026033107425852900_vzaf100-B7","doi-asserted-by":"crossref","first-page":"81","DOI":"10.1007\/s00403-019-01972-3","article-title":"The IL-4\/IL-13 axis in skin fibrosis and scarring: mechanistic concepts and therapeutic targets","volume":"312","author":"Nguyen","year":"2020","journal-title":"Arch Dermatol Res"},{"key":"2026033107425852900_vzaf100-B8","doi-asserted-by":"crossref","first-page":"2051","DOI":"10.4049\/jimmunol.1501491","article-title":"CD4+ group 1 innate lymphoid cells form a functionally distinct subset that is increased in systemic sclerosis","volume":"196","author":"Roan","year":"2016","journal-title":"J Immunol"}],"container-title":["Skin Health and Disease"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/skinhd\/article-pdf\/6\/2\/160\/66326310\/vzaf100.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/skinhd\/article-pdf\/6\/2\/160\/66326310\/vzaf100.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T11:43:04Z","timestamp":1774957384000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/skinhd\/article\/6\/2\/160\/8418302"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,9]]},"references-count":8,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2026,1,9]]},"published-print":{"date-parts":[[2026,3,31]]}},"URL":"https:\/\/doi.org\/10.1093\/skinhd\/vzaf100","relation":{},"ISSN":["2690-442X"],"issn-type":[{"value":"2690-442X","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2026,3]]},"published":{"date-parts":[[2026,1,9]]}}}