{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,21]],"date-time":"2026-09-21T15:22:25Z","timestamp":1790004145735,"version":"4.0.1"},"reference-count":13,"publisher":"BMJ","issue":"5","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2025,4,22]]},"published-print":{"date-parts":[[2025,5]]},"abstract":"<jats:p>This case report presents a woman in her 20s with VACTERL association who presented with acute on chronic 10-year pelvic pain and haematometra. Initially, diagnostic imaging suspected a didelphic uterus with a dilated right horn on CT imaging. However, after physical examination and further imaging, further diagnosis of unicornuate uterus with non-communicating horn was noted. After years of expectant management, the patient began to experience severe malnutrition and metabolic derangements. After significant patient-centred discussions regarding pain relief, the patient was refractory to oral contraceptive pills and later started on GnRH antagonists. After the patient underwent an abdominal hysterectomy and right salpingectomy with resolution of pain symptoms. The case highlights the complexities of managing patients with multiple congenital anomalies, particularly VACTERL, and while not in the constellation of syndromes, gynaecological complications may arise due to common embryological origins. This case emphasises the importance of a multidisciplinary approach in challenging scenarios.<\/jats:p>","DOI":"10.1136\/bcr-2024-262730","type":"journal-article","created":{"date-parts":[[2025,5,2]],"date-time":"2025-05-02T07:44:58Z","timestamp":1746171898000},"page":"e262730","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":2,"title":["Management of VACTERL association with uterine anomaly: complex acute on chronic 10-year pelvic pain with haematometra"],"prefix":"10.1136","volume":"18","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3586-7953","authenticated-orcid":false,"given":"Richard","family":"Hsu","sequence":"first","affiliation":[{"name":"Department of Obestetrics and Gynecology, Wayne State University School of Medicine, Detroit, MI, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Urvashi","family":"Gupta","sequence":"additional","affiliation":[{"name":"Department of Obestetrics and Gynecology, Wayne State University School of Medicine, Detroit, MI, USA"},{"name":"Department of Obstetrics and Gynecology, Detroit Medical Center, Detroit, MI, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bria","family":"Elrod","sequence":"additional","affiliation":[{"name":"Department of Obestetrics and Gynecology, Wayne State University School of Medicine, Detroit, MI, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Patrice","family":"Harold","sequence":"additional","affiliation":[{"name":"Department of Obestetrics and Gynecology, Wayne State University School of Medicine, Detroit, MI, USA"},{"name":"Department of Obstetrics and Gynecology, Detroit Medical Center, Detroit, MI, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2025,5,2]]},"reference":[{"key":"2025082804253796000_18.5.e262730.1","doi-asserted-by":"crossref","first-page":"1715","DOI":"10.1038\/ejhg.2016.86","article-title":"Copy number variations in 375 patients with VACTERL association","volume":"24","author":"Brosens","year":"2016","journal-title":"Eur J Hum Genet"},{"key":"2025082804253796000_18.5.e262730.2","first-page":"105857","article-title":"Surgical management of an obstructive M\u00fcllerian anomaly in a patient with anorectal malformation","volume":"82","author":"Theodorou","year":"2021","journal-title":"Int J Surg Case Rep"},{"key":"2025082804253796000_18.5.e262730.3","doi-asserted-by":"crossref","first-page":"71","DOI":"10.1186\/s12905-019-0768-4","article-title":"Unicornuate uterus with a rudimentary non-communicating cavitary horn in association with VACTERL association: case report","volume":"19","author":"Obeidat","year":"2019","journal-title":"BMC Womens Health"},{"key":"2025082804253796000_18.5.e262730.4","doi-asserted-by":"crossref","unstructured":"Santana Gonz\u00e1lez L , Artibani M , Ahmed AA . 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In: Early pregnancy. 90.2017.","DOI":"10.1017\/9781139976787.010"},{"key":"2025082804253796000_18.5.e262730.7","doi-asserted-by":"publisher","DOI":"10.1148\/radiol.2331020777"},{"key":"2025082804253796000_18.5.e262730.8","doi-asserted-by":"crossref","first-page":"195","DOI":"10.1016\/j.jpurol.2018.01.011","article-title":"Screening for Mullerian anomalies in patients with unilateral renal agenesis: Leveraging early detection to prevent complications","volume":"14","author":"Friedman","year":"2018","journal-title":"J Pediatr Urol"},{"key":"2025082804253796000_18.5.e262730.9","doi-asserted-by":"crossref","first-page":"1238","DOI":"10.1016\/j.fertnstert.2021.09.025","article-title":"ASRM m\u00fcllerian anomalies classification 2021","volume":"116","year":"2021","journal-title":"Fertil Steril"},{"key":"2025082804253796000_18.5.e262730.10","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejmg.2007.04.002"},{"key":"2025082804253796000_18.5.e262730.11","doi-asserted-by":"publisher","DOI":"10.1002\/bdra.23302"},{"key":"2025082804253796000_18.5.e262730.12","unstructured":"Acker SN , et al . Does presence of a VACTERL anomaly predict an associated gynecologic anomaly in patients with anorectal malformation. 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