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The median age of death is 32\u00a0years and 60% of XP patients die before the age of 20\u00a0years. cSCC in patients with XP exhibits an exceptionally high mutation burden, suggesting a favorable response to immune checkpoint inhibitors (ICIs). We present the case of a 7\u2010year\u2010old boy with XP and a large facial cSCC complicated by cervical lymph node metastases. Following a tumor board recommendation, systemic immunotherapy with cemiplimab was initiated. Following therapy, the tumors rapidly and completely regressed. To date, only 10 XP patients worldwide have been reported to receive ICIs for inoperable and\/or advanced cSCC, with all cases demonstrating tumor regression under ICI treatment. Among these, three were pediatric cases with XP\u2010C (one 7\u2010year\u2010old and two 6\u2010year\u2010old children), one of whom had sarcomatoid cSCC. Incidence and nature of adverse events in XP patients were comparable to those observed in the general population. In line with the previously reported ICI\u2010treated XP children, the present case confirms that anti\u2010PD\u20101 inhibitors are highly effective in children with XP and advanced cSCC.<\/jats:p>","DOI":"10.1111\/ddg.15648","type":"journal-article","created":{"date-parts":[[2025,1,27]],"date-time":"2025-01-27T07:59:07Z","timestamp":1737964747000},"page":"303-308","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Immune checkpoint inhibitors for children with xeroderma pigmentosum and advanced cutaneous squamous cell carcinoma: A case presentation and brief review"],"prefix":"10.1111","volume":"23","author":[{"given":"Thilo","family":"Gambichler","sequence":"first","affiliation":[{"name":"Department of Dermatology Dortmund Hospital University Witten\u2010Herdecke Faculty of Health\/School of Medicine  Dortmund Germany"},{"name":"Department of Dermatology Cristian Hospital Unna  Unna Germany"},{"name":"Department of Dermatology Ruhr\u2010University Bochum  Bochum Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Julia","family":"Hyun","sequence":"additional","affiliation":[{"name":"Department of Dermatology Venereology and Allergology Helios St. Johannes Hospital Duisburg  Duisburg Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Frank","family":"Oellig","sequence":"additional","affiliation":[{"name":"Department of Pathology M\u00fclheim an der Ruhr, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"J\u00fcrgen C.","family":"Becker","sequence":"additional","affiliation":[{"name":"Translational Skin Cancer Research DKTK Partner Site Essen\/D\u00fcsseldorf, Germany"},{"name":"West German Cancer Center Dermatology University Duisburg\u2010Essen  Essen Germany"},{"name":"German Cancer Research Center (DKFZ)  Heidelberg 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