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We report the case of a middle-aged man with multiple bilateral eyelid hidrocystomas, palmoplantar keratoderma, hypodontia and nail dystrophy, in keeping with a diagnosis of Sch\u00f6pf-Schulz-Passarge syndrome, an exceedingly rare ectodermal dysplasia. Hyperprolactinaemia was found on testing, and radiology revealed an incidental prolactinoma. Raised serum prolactin has not been previously associated with Sch\u00f6pf-Schulz-Passarge syndrome. The significance of raised serum prolactin in the context of multiple eyelid hidrocystomas is discussed.<\/jats:p>","DOI":"10.1136\/bcr-2025-267144","type":"journal-article","created":{"date-parts":[[2026,6,26]],"date-time":"2026-06-26T13:10:57Z","timestamp":1782479457000},"page":"e267144","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Pituitary adenoma-associated with Sch\u00f6pf-Schulz-Passarge syndrome"],"prefix":"10.1136","volume":"19","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5839-8566","authenticated-orcid":false,"given":"Robert Thomas","family":"Brady","sequence":"first","affiliation":[{"name":"Ophthalmology, Addenbrooke\u2019s Hospital, Cambridge, England, UK"},{"name":"Adnexal, Moorfields Eye Hospital, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kieren","family":"Allinson","sequence":"additional","affiliation":[{"name":"Pathology, Addenbrooke\u2019s Hospital, Cambridge, England, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2783-641X","authenticated-orcid":false,"given":"Cornelius","family":"Ren\u00e9","sequence":"additional","affiliation":[{"name":"Ophthalmology, Addenbrooke\u2019s Hospital, Cambridge, England, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2026,6,26]]},"reference":[{"key":"2026062606101441000_19.6.e267144.1","first-page":"57","article-title":"Hidrocystomas--a brief review","volume":"8","author":"Sarabi","year":"2006","journal-title":"MedGenMed"},{"key":"2026062606101441000_19.6.e267144.2","doi-asserted-by":"publisher","DOI":"10.1159\/000246098"},{"key":"2026062606101441000_19.6.e267144.3","doi-asserted-by":"crossref","first-page":"e96","DOI":"10.1097\/IOP.0000000000002855","article-title":"Multiple Eyelid Apocrine Hidrocystomas in a Patient With Prolactinoma","volume":"41","author":"Akgoz","year":"2025","journal-title":"Ophthalmic Plast Reconstr Surg"},{"key":"2026062606101441000_19.6.e267144.4","doi-asserted-by":"crossref","first-page":"e83","DOI":"10.1097\/IOP.0000000000001069","article-title":"Multiple Eyelid Cysts (Apocrine and Eccrine Hidrocystomas, Trichilemmal Cyst, and Hybrid Cyst) in a Patient With a Prolactinoma","volume":"34","author":"Ma","year":"2018","journal-title":"Ophthalmic Plast Reconstr Surg"},{"key":"2026062606101441000_19.6.e267144.5","first-page":"219","article-title":"Syndrome of cystic eyelids, palmo-plantar keratosis, hypodontia and hypotrichosis as a possible autosomal recessive trait","volume":"7","author":"Sch\u00f6pf","year":"1971","journal-title":"Birth Defects Orig Artic Ser"},{"key":"2026062606101441000_19.6.e267144.6","doi-asserted-by":"publisher","DOI":"10.1001\/archderm.1962.01590120006002"},{"key":"2026062606101441000_19.6.e267144.7","doi-asserted-by":"publisher","DOI":"10.1016\/j.diff.2025.100838"},{"key":"2026062606101441000_19.6.e267144.8","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2009.06.001"},{"key":"2026062606101441000_19.6.e267144.9","doi-asserted-by":"crossref","first-page":"1063","DOI":"10.1038\/ejhg.2013.300","article-title":"Variability in dentofacial phenotypes in four families with WNT10A mutations","volume":"22","author":"Vink","year":"2014","journal-title":"Eur J Hum Genet"},{"key":"2026062606101441000_19.6.e267144.10","doi-asserted-by":"crossref","first-page":"3","DOI":"10.1038\/s41392-021-00762-6","article-title":"Wnt\/\u03b2-catenin signalling: function, biological mechanisms, and therapeutic opportunities","volume":"7","author":"Liu","year":"2022","journal-title":"Sig Transduct Target Ther"},{"key":"2026062606101441000_19.6.e267144.11","doi-asserted-by":"crossref","DOI":"10.3389\/fonc.2020.566637","article-title":"Comparative Assessment of the WNT\/\u03b2-Catenin Pathway, CacyBP\/SIP, and the Immunoproteasome Subunit LMP7 in Various Histological Types of Renal Cell Carcinoma","volume":"10","author":"Piotrowska","year":"2020","journal-title":"Front Oncol"},{"key":"2026062606101441000_19.6.e267144.12","doi-asserted-by":"publisher","DOI":"10.3892\/ol.2019.10035"},{"key":"2026062606101441000_19.6.e267144.13","doi-asserted-by":"publisher","DOI":"10.3892\/or.2017.5777"},{"key":"2026062606101441000_19.6.e267144.14","doi-asserted-by":"crossref","first-page":"607","DOI":"10.2340\/00015555-0547","article-title":"Sch\u00f6pf-Schulz-Passarge syndrome: further delineation of the phenotype and genetic considerations","volume":"88","author":"Castori","year":"2008","journal-title":"Acta Derm Venereol"},{"key":"2026062606101441000_19.6.e267144.15","first-page":"809","article-title":"Multiple eccrine hydrocystomas of the eyelids in the framework of Sch\u00f6pf syndrome. A case report","volume":"23","author":"Dot","year":"2000","journal-title":"J Fr Ophtalmol"},{"key":"2026062606101441000_19.6.e267144.16","doi-asserted-by":"publisher","DOI":"10.1016\/S0303-7207(02)00286-1"},{"key":"2026062606101441000_19.6.e267144.17","doi-asserted-by":"crossref","DOI":"10.3390\/ijms25137100","article-title":"Prolactin: A Mammalian Stress Hormone and Its Role in Cutaneous Pathophysiology","volume":"25","author":"Langan","year":"2024","journal-title":"Int J Mol Sci"},{"key":"2026062606101441000_19.6.e267144.18","doi-asserted-by":"publisher","DOI":"10.1016\/j.tem.2010.06.001"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2025-267144","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,26]],"date-time":"2026-06-26T13:10:59Z","timestamp":1782479459000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2025-267144"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6]]},"references-count":18,"journal-issue":{"issue":"6","published-online":{"date-parts":[[2026,6,26]]},"published-print":{"date-parts":[[2026,6]]}},"alternative-id":["10.1136\/bcr-2025-267144"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2025-267144","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,6]]}}}