{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,4]],"date-time":"2026-09-04T16:21:43Z","timestamp":1788538903247,"version":"build-2803163510"},"reference-count":10,"publisher":"Ovid Technologies (Wolters Kluwer Health)","content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Herlyn\u2013Werner\u2013Wunderlich syndrome, also known as obstructed hemivagina and ipsilateral renal anomaly (OHVIRA) syndrome, is a rare congenital M\u00fcllerian duct anomaly, occurring in approximately 1 in 1,000,000 females. It is characterised by a triad of uterine didelphys, obstructed hemivagina and ipsilateral renal agenesis. Patients typically present with chronic pelvic pain, dysmenorrhea and endometriosis, which can lead to diagnostic bias and the misattribution of all pelvic pain to their underlying gynaecological condition. We present the case of a 19-year-old female with a known diagnosis of OHVIRA syndrome who presented with acute right iliac fossa (RIF) pain and raised inflammatory markers. Despite her complex gynaecological background, imaging confirmed a diagnosis of acute appendicitis with impending perforation. She underwent a successful laparoscopic appendicectomy. The patient had experienced multiple prior episodes of RIF pain, previously attributed to her gynaecological condition, without further surgical evaluation. This case underscores the importance of maintaining a broad differential diagnosis in patients with complex gynaecological anomalies and highlights the risk of diagnostic overshadowing. A comprehensive assessment for acute surgical pathology should always be considered in this cohort to avoid delays in diagnosis and treatment.<\/jats:p>","DOI":"10.4103\/jmas.jmas_538_25","type":"journal-article","created":{"date-parts":[[2026,9,4]],"date-time":"2026-09-04T16:01:03Z","timestamp":1788537663000},"update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":0,"title":["Acute appendicitis in a patient with obstructed hemivagina and ipsilateral renal anomaly syndrome: Diagnostic overshadowing in recurrent pelvic pain"],"prefix":"10.4103","author":[{"given":"Rafique Umer","family":"Harvitkar","sequence":"first","affiliation":[{"name":"Department of General Surgery, St Richards Hospital, Chichester, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Neriah","family":"Mangion","sequence":"additional","affiliation":[{"name":"Foundation Year 2, Royal Sussex County Hospital, Kent Surrey Sussex, Brighton, England"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ioannis","family":"Hannadjas","sequence":"additional","affiliation":[{"name":"Department of General Surgery, Brighton and Sussex University Hospitals, Kent Surrey Sussex, Brighton, England"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seshu Kumar","family":"Bylapudi","sequence":"additional","affiliation":[{"name":"Department of General Surgery, Leicester Royal Infirmary, Leicester, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nida","family":"Khan","sequence":"additional","affiliation":[{"name":"Department of General Surgery, Royal Sussex County Hospital, Brighton, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2026,9,3]]},"reference":[{"issue":"Suppl 3","key":"R1-20260904","first-page":"136","article-title":"Herlyn-Werner-Wunderlich syndrome in a young female mimicking acute appendicitis","volume":"109","author":"Fiechter","year":"2022","journal-title":"Br J Surg"},{"key":"R2-20260904","doi-asserted-by":"crossref","first-page":"105911","DOI":"10.1016\/j.ijscr.2021.105911","article-title":"Acute abdomen: Unusual presentation of Herlyn-Werner Wunderlich syndrome","volume":"82","author":"Saleh","year":"2021","journal-title":"Int J Surg Case Rep"},{"key":"R3-20260904","first-page":"275","article-title":"OHVIRA syndrome presenting with acute abdomen findings treated with minimally invasive method: Three case reports","volume":"122","author":"G\u00fcnd\u00fcz","year":"2022","journal-title":"Acta Chir Belg"},{"key":"R4-20260904","doi-asserted-by":"crossref","first-page":"562","DOI":"10.31729\/jnma.7444","article-title":"Obstructed hemivagina and ipsilateral renal agenesis syndrome: A case report","volume":"60","author":"Chapagain","year":"2022","journal-title":"J Nepal Med Assoc"},{"key":"R5-20260904","first-page":"136","article-title":"Herlyn Werner Wunderlich syndrome with hematocolpos: An unusual case report of full diagnostic approach and treatment","volume":"10","author":"Bhoil","year":"2016","journal-title":"Int J Fertil Steril"},{"key":"R6-20260904","first-page":"165","article-title":"Herlyn-Werner-Wunderlich syndrome in a multiparous female","volume":"7","author":"Sidhu","year":"2021","journal-title":"BJR Case Rep"},{"key":"R7-20260904","doi-asserted-by":"crossref","first-page":"131","DOI":"10.1177\/2010105818797141","article-title":"Herlyn-Werner-Wunderlich syndrome: Case report of a newborn presenting with interlabial cyst","volume":"28","author":"Cheong","year":"2019","journal-title":"Proc Singap Healthc"},{"key":"R8-20260904","doi-asserted-by":"crossref","first-page":"657","DOI":"10.1007\/s00247-007-0497-y","article-title":"Herlyn-Werner-Wunderlich syndrome: Uterus didelphys, blind hemivagina and ipsilateral renal agenesis. Sonographic and MR findings in 11 cases","volume":"37","author":"Orazi","year":"2007","journal-title":"Pediatr Radiol"},{"key":"R9-20260904","doi-asserted-by":"crossref","first-page":"357","DOI":"10.1016\/j.clinimag.2013.12.011","article-title":"OHVIRA syndrome presenting with acute abdomen: A case report and review of the literature","volume":"38","author":"Gungor","year":"2014","journal-title":"Clin Imaging"},{"key":"R10-20260904","doi-asserted-by":"crossref","first-page":"e00185","DOI":"10.1016\/j.crwh.2020.e00185","article-title":"Right-sided obstructed hemivagina ipsilateral renal agenesis (OHVIRA): A case report","volume":"26","author":"Malarazhagan","year":"2020","journal-title":"Case Rep Womens Health"}],"container-title":["Journal of Minimal Access Surgery"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.4103\/jmas.jmas_538_25","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,4]],"date-time":"2026-09-04T16:01:04Z","timestamp":1788537664000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.4103\/jmas.jmas_538_25"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,9,3]]},"references-count":10,"URL":"https:\/\/doi.org\/10.4103\/jmas.jmas_538_25","relation":{},"ISSN":["0972-9941","1998-3921"],"issn-type":[{"value":"0972-9941","type":"print"},{"value":"1998-3921","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,9,3]]},"assertion":[{"value":"2025-12-05","name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-04-27","name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}}]}}