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The origin of these lesions is unclear, but their appearance seems to be associated with various forms of androgen excess, including androgen therapy for transgender treatment or disorders of sex development, such as classic congenital adrenal hyperplasia (CAH). This is the first described case of ectopic prostate tissue in the cervix uteri of a 46,XX patient with a confirmed diagnosis of non-classic CAH due to 21-OHD and a history of mild adrenal androgen excess. Case presentation: We describe a 34-year-old patient with a genetic diagnosis of non-classic CAH due to 21-hydroxylase deficiency (21-OHD) with a female karyo- and phenotype and a history of mild adrenal androgen excess. Due to dysplasia in the cervical smear, conization had to be performed, revealing ectopic prostate tissue in the cervix uteri of the patient. Conclusions: An association between androgen excess and the occurrence of prostate tissue is likely and should therefore be considered as a differential diagnosis for atypical tissue in the female genital tract.<\/jats:p>","DOI":"10.3390\/jcm11154307","type":"journal-article","created":{"date-parts":[[2022,7,25]],"date-time":"2022-07-25T21:20:59Z","timestamp":1658784059000},"page":"4307","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Ectopic Prostate Tissue in the Uterine Cervix of a Female with Non-Classic Congenital Adrenal Hyperplasia\u2014A Case Report"],"prefix":"10.3390","volume":"11","author":[{"given":"Lea","family":"Tschaidse","sequence":"first","affiliation":[{"name":"Medizinische Klinik und Poliklinik IV, Klinikum der Universit\u00e4t M\u00fcnchen, LMU M\u00fcnchen, 80336 Munich, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Matthias K.","family":"Auer","sequence":"additional","affiliation":[{"name":"Medizinische Klinik und Poliklinik IV, Klinikum der Universit\u00e4t M\u00fcnchen, LMU M\u00fcnchen, 80336 Munich, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ilja","family":"Dubinski","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology, Dr. von Haunersches Children\u2019s Hospital, Klinikum der Universit\u00e4t M\u00fcnchen, LMU M\u00fcnchen, 80336 Munich, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1770-4392","authenticated-orcid":false,"given":"Christian","family":"Lottspeich","sequence":"additional","affiliation":[{"name":"Medizinische Klinik und Poliklinik IV, Klinikum der Universit\u00e4t M\u00fcnchen, LMU M\u00fcnchen, 80336 Munich, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hanna","family":"Nowotny","sequence":"additional","affiliation":[{"name":"Medizinische Klinik und Poliklinik IV, Klinikum der Universit\u00e4t M\u00fcnchen, LMU M\u00fcnchen, 80336 Munich, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Heinrich","family":"Schmidt","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology, Dr. von Haunersches Children\u2019s Hospital, Klinikum der Universit\u00e4t M\u00fcnchen, LMU M\u00fcnchen, 80336 Munich, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nadezda","family":"Gut","sequence":"additional","affiliation":[{"name":"Zentrum f\u00fcr Pathologie Allg\u00e4u (ZfPA), Medizinisches Versorgungzentrum am Klinikum Kempten, 87439 Kempten, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nicole","family":"Reisch","sequence":"additional","affiliation":[{"name":"Medizinische Klinik und Poliklinik IV, Klinikum der Universit\u00e4t M\u00fcnchen, LMU M\u00fcnchen, 80336 Munich, Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1968","published-online":{"date-parts":[[2022,7,25]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"1040","DOI":"10.1097\/PAS.0000000000001486","article-title":"Prostatic Metaplasia of the Vagina and Uterine Cervix: An Androgen-associated Glandular Lesion of Surface Squamous Epithelium","volume":"44","author":"Anderson","year":"2020","journal-title":"Am. 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