{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,25]],"date-time":"2026-04-25T07:05:07Z","timestamp":1777100707405,"version":"3.51.4"},"reference-count":9,"publisher":"BMJ","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Reports"],"accepted":{"date-parts":[[2018,9,12]]},"abstract":"<jats:p>A 42-year-old African man presented with hypogonadic phenotypical features, including gynoid body distribution, gynaecomastia, absent facial and truncal hair and micropenis. He denied ever experiencing development of male secondary sex characteristics. Endocrine testing revealed hypergonadotropic hypogonadism and undetectable AMH. Human chorionic gonadotropin (hCG) stimulation test failed to increase testosterone levels. Peripheral blood karyotype was 46, XY. Clinical examination and abdominal\/pelvic\/scrotal ultrasound and MRI failed to identify any testicular structures\/remnants. Given the clinical course and the biochemical\u2013radiological presentation, the diagnosis of bilateral anorchia was made (after more than four decades of its probable onset), and surgical exploration was decided against. The patient was subsequently started on monthly intramuscular testosterone experiencing progressive normal virilisation.<\/jats:p>","DOI":"10.1136\/bcr-2018-225530","type":"journal-article","created":{"date-parts":[[2018,10,13]],"date-time":"2018-10-13T01:45:33Z","timestamp":1539395133000},"page":"bcr-2018-225530","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":3,"title":["Case of newly diagnosed bilateral anorchia in a 42-year-old male patient"],"prefix":"10.1136","volume":"2018","author":[{"given":"Francisco","family":"Sousa Santos","sequence":"first","affiliation":[{"name":"Endocrinology, Hospital de Egas Moniz, Lisbon, Portugal"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"C\u00e1tia","family":"Ferrinho","sequence":"additional","affiliation":[{"name":"Endocrinology, Hospital de Egas Moniz, Lisbon, Portugal"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Clotilde","family":"Limbert","sequence":"additional","affiliation":[{"name":"Endocrinology, Hospital de Egas Moniz, Lisbon, Portugal"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Carlos","family":"Vasconcelos","sequence":"additional","affiliation":[{"name":"Endocrinology, Hospital de Egas Moniz, Lisbon, Portugal"},{"name":"Endocrinology, Hospital de Egas Moniz, Lisbon, Portugal"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2018,10,12]]},"reference":[{"key":"2025082723324192000_2018.oct12_1.bcr-2018-225530.1","doi-asserted-by":"crossref","first-page":"116","DOI":"10.4274\/Jcrpe.728","article-title":"Vanishing testes: a literature review","volume":"4","author":"Pirgon","year":"2012","journal-title":"J Clin Res Pediatr Endocrinol"},{"key":"2025082723324192000_2018.oct12_1.bcr-2018-225530.2","doi-asserted-by":"crossref","first-page":"e23292","DOI":"10.1371\/journal.pone.0023292","article-title":"Clinical, biological and genetic analysis of anorchia in 26 boys","volume":"6","author":"Brauner","year":"2011","journal-title":"PLoS One"},{"key":"2025082723324192000_2018.oct12_1.bcr-2018-225530.3","doi-asserted-by":"crossref","first-page":"206","DOI":"10.1016\/j.jpurol.2006.08.007","article-title":"Natural history of testicular regression syndrome and consequences for clinical management","volume":"3","author":"Hegarty","year":"2007","journal-title":"J Pediatr Urol"},{"key":"2025082723324192000_2018.oct12_1.bcr-2018-225530.4","doi-asserted-by":"crossref","first-page":"151.e1","DOI":"10.1016\/j.jpurol.2015.01.018","article-title":"Is routine excision of testicular remnants in testicular regression syndrome indicated?","volume":"11","author":"Nataraja","year":"2015","journal-title":"J Pediatr Urol"},{"key":"2025082723324192000_2018.oct12_1.bcr-2018-225530.5","doi-asserted-by":"crossref","first-page":"65","DOI":"10.1159\/000474420","article-title":"The vanishing testis: anatomical and histological findings","volume":"31","author":"Merry","year":"1997","journal-title":"Eur Urol"},{"key":"2025082723324192000_2018.oct12_1.bcr-2018-225530.6","doi-asserted-by":"crossref","unstructured":"Nataraja RM , Yeap E , Healy CJ , et al . Presence of viable germ cells in testicular regression syndrome remnants: Is routine excision indicated? A systematic review. Pediatr Surg Int 2018;34.doi:10.1007\/s00383-017-4206-0","DOI":"10.1007\/s00383-017-4206-0"},{"key":"2025082723324192000_2018.oct12_1.bcr-2018-225530.7","doi-asserted-by":"crossref","first-page":"712","DOI":"10.1016\/S0022-5347(01)66507-8","article-title":"The remnant orchiectomy","volume":"155","author":"Rozanski","year":"1996","journal-title":"J Urol"},{"key":"2025082723324192000_2018.oct12_1.bcr-2018-225530.8","doi-asserted-by":"publisher","DOI":"10.1016\/j.juro.2009.04.032"},{"key":"2025082723324192000_2018.oct12_1.bcr-2018-225530.9","doi-asserted-by":"crossref","first-page":"79","DOI":"10.1590\/2359-3997000000127","article-title":"Vanishing testes syndrome-related osteoporosis and high cardio-metabolic risk in an adult male with long term untreated hypergonadotropic hypogonadism","volume":"60","author":"Carsote","year":"2016","journal-title":"Arch Endocrinol Metab"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2018-225530","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T06:32:51Z","timestamp":1756362771000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2018-225530"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,10,12]]},"references-count":9,"alternative-id":["10.1136\/bcr-2018-225530"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2018-225530","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,10,12]]}}}