{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,28]],"date-time":"2026-07-28T01:58:40Z","timestamp":1785203920302,"version":"3.55.0"},"reference-count":11,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T00:00:00Z","timestamp":1785110400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T00:00:00Z","timestamp":1785110400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Dermatology"],"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Harlequin ichthyosis (HI) is a rare epidermal differentiation disorder characterized by thick hyperkeratotic plates, fissuring, and high neonatal morbidity. Long\u2010term management remains challenging. We report two children with genetically confirmed HI who demonstrated meaningful improvement in pruritus, erythema, scaling, and overall well\u2010being following dupilumab therapy, suggesting a potential role for IL\u20104\/IL\u201013 blockade as an adjunct or alternative treatment in HI.<\/jats:p>","DOI":"10.1111\/pde.70327","type":"journal-article","created":{"date-parts":[[2026,7,28]],"date-time":"2026-07-28T01:02:37Z","timestamp":1785200557000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Dupilumab Provides Meaningful Clinical Responses in Harlequin Ichthyosis (\n                    <i>ABCA12<\/i>\n                    \u2010\n                    <scp>nEDD<\/scp>\n                    ): A Report of Two Cases"],"prefix":"10.1111","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3078-1146","authenticated-orcid":false,"given":"Minu Jose","family":"Chiramel","sequence":"first","affiliation":[{"name":"Department of Pediatric Dermatology The Hospital for Sick Children  Toronto Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0100-1801","authenticated-orcid":false,"given":"Cynthia","family":"Chen","sequence":"additional","affiliation":[{"name":"Washington University School of Medicine  St Louis Missouri USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elaine","family":"Siegfried","sequence":"additional","affiliation":[{"name":"Division of Pediatric Dermatology, SSM Health\/Cardinal, Glennon Children's Hospital, Departments of Pediatrics and Dermatology St Louis University School of Medicine  Missouri USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5407-3972","authenticated-orcid":false,"given":"Rebecca","family":"Levy","sequence":"additional","affiliation":[{"name":"Department of Pediatric Dermatology The Hospital for Sick Children  Toronto Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,7,27]]},"reference":[{"issue":"6","key":"e_1_2_7_2_1","doi-asserted-by":"crossref","DOI":"10.3390\/children9060893","article-title":"Management of Harlequin Ichthyosis: A Brief Review of the Recent Literature","volume":"9","author":"Tsivilika M.","year":"2022","journal-title":"Children"},{"issue":"4","key":"e_1_2_7_3_1","doi-asserted-by":"crossref","DOI":"10.3390\/biom13040634","article-title":"Dupilumab in Inflammatory Skin Diseases: A Systematic Review","volume":"13","author":"Olbrich H.","year":"2023","journal-title":"Biomolecules"},{"issue":"3","key":"e_1_2_7_4_1","doi-asserted-by":"crossref","DOI":"10.1016\/j.jid.2025.02.082","article-title":"74: Dupilumab Reduces Itch and Pain in Pruritic Genetic Skin Disorders: Interim Analysis From a Single\u2010Site Study","volume":"145","author":"Gallagher K.","year":"2025","journal-title":"Journal of Investigative Dermatology"},{"issue":"1","key":"e_1_2_7_5_1","doi-asserted-by":"crossref","first-page":"94","DOI":"10.5468\/ogs.2020.63.1.94","article-title":"Prenatal Diagnosis of Harlequin Ichthyosis: A Case Report","volume":"63","author":"Vijayakumari M.","year":"2020","journal-title":"Obstetrics & Gynecology Science"},{"issue":"1","key":"e_1_2_7_6_1","doi-asserted-by":"crossref","first-page":"155","DOI":"10.1016\/j.jaci.2018.08.022","article-title":"Dupilumab Progressively Improves Systemic and Cutaneous Abnormalities in Patients With Atopic Dermatitis","volume":"143","author":"Guttman\u2010Yassky E.","year":"2019","journal-title":"Journal of Allergy and Clinical Immunology"},{"issue":"2","key":"e_1_2_7_7_1","doi-asserted-by":"crossref","first-page":"436","DOI":"10.1093\/hmg\/ddu459","article-title":"Fetal Inhibition of Inflammation Improves Disease Phenotypes in Harlequin Ichthyosis","volume":"24","author":"Cottle D. L.","year":"2015","journal-title":"Human Molecular Genetics"},{"issue":"2","key":"e_1_2_7_8_1","doi-asserted-by":"crossref","first-page":"185","DOI":"10.1111\/ajd.14197","article-title":"Biologics for Inherited Disorders of Keratinisation: A Systematic Review","volume":"65","author":"Chen M. K. Y.","year":"2024","journal-title":"Australasian Journal of Dermatology"},{"issue":"2","key":"e_1_2_7_9_1","doi-asserted-by":"crossref","first-page":"305","DOI":"10.1007\/s00403-022-02325-3","article-title":"Secukinumab Responses Vary Across the Spectrum of Congenital Ichthyosis in Adults","volume":"315","author":"Lefferdink R.","year":"2023","journal-title":"Archives of Dermatological Research"},{"issue":"3","key":"e_1_2_7_10_1","doi-asserted-by":"crossref","first-page":"535","DOI":"10.1016\/j.jid.2018.08.027","article-title":"Profiling Immune Expression to Consider Repurposing Therapeutics for the Ichthyoses","volume":"139","author":"Paller A. S.","year":"2019","journal-title":"Journal of Investigative Dermatology"},{"issue":"1","key":"e_1_2_7_11_1","doi-asserted-by":"crossref","first-page":"152","DOI":"10.1016\/j.jaci.2016.07.019","article-title":"An IL\u201017\u2010Dominant Immune Profile Is Shared Across the Major Orphan Forms of Ichthyosis","volume":"139","author":"Paller A. S.","year":"2017","journal-title":"Journal of Allergy and Clinical Immunology"},{"issue":"10","key":"e_1_2_7_12_1","doi-asserted-by":"crossref","first-page":"2157","DOI":"10.1016\/j.jid.2018.03.1523","article-title":"The Major Orphan Forms of Ichthyosis Are Characterized by Systemic T\u2010Cell Activation and Th\u201017\/Tc\u201017\/Th\u201022\/Tc\u201022 Polarization in Blood","volume":"138","author":"Czarnowicki T.","year":"2018","journal-title":"Journal of Investigative Dermatology"}],"container-title":["Pediatric Dermatology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/pde.70327","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/pde.70327","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/pde.70327","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,28]],"date-time":"2026-07-28T01:02:39Z","timestamp":1785200559000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/pde.70327"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,7,27]]},"references-count":11,"alternative-id":["10.1111\/pde.70327"],"URL":"https:\/\/doi.org\/10.1111\/pde.70327","archive":["Portico"],"relation":{},"ISSN":["0736-8046","1525-1470"],"issn-type":[{"value":"0736-8046","type":"print"},{"value":"1525-1470","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,7,27]]},"assertion":[{"value":"2026-02-15","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-20","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-27","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"pde.70327"}}