Uterine rudimentary horn and ovarian endometriosis with a special emphasis on VACTERL association: a report of two cases

Authors

  • Arushi Bhadwal Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi
  • Sonam Sharma Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi
  • Sunil Ranga Department of Pathology, Vardhman Mahavir Medical College and Safdarjung Hospital, New Delhi

DOI:

https://doi.org/10.18203/2320-1770.ijrcog20241793

Keywords:

Mullerian duct anomalies, Unicornuate uterus, Rudimentary uterine horn, Endometriosis, VACTERL

Abstract

Mullerian duct anomalies are a wide and complex spectrum of congenital abnormalities that are often associated with renal as well as axial skeletal anomalies or can be a part of multiple malformation syndrome and may cause numerous other gynecological complications. We herein describe two young girls who presented with a common complaint of abdominal pain and had normal secondary sexual characteristics.The first case had a history of imperforate anus, and urinary incontinence due to renal anomalies including ectopic kidney, renal agenesis, duplex ureter along with sacral agenesis which were confirmed on imaging. VACTERL (V-vertebral anomalies, A-anorectal malformations, C-cardiovascular anomalies, T- tracheoesophageal fistula, E-esophageal atresia, R-renal anomalies, and L-limb defects) association was considered. To the best of our knowledge, this is the fourth such case with concomitant VACTERL association. Surgical resections were performed in both cases, after clinical-radiological correlation. Histopathological examination of specimens revealed a unicornuate uterus with a rudimentary horn and ovarian endometriosis. In conclusion, early identification and prompt intervention through a multidisciplinary approach are paramount for managing such anomalies as they can have many detrimental implications.

 

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References

Sharma S. A rare incidental case of an accessory fallopian tube. Fertil Sci Res. 2020;7(1):117-20.

Caserta D, Mallozzi M, Meldolesi C, Bianchi P, Moscarini M. Pregnancy in a unicornuate uterus: a case report. J Med Case Rep. 2014;8:130.

Li S, Qayyum A, Coakley FV, Hricak H. Association of renal agenesis and mullerian duct anomalies. J Comput Assist Tomogr. 2000;24(6):829-34.

Osman NM, Hamza AM, Elamin HAM. Congenital vaginal obstruction in a newborn. Sudan J Paediatr. 2019;19(2):145-8.

Messina M, Severi FM, Bocchi C, Ferrucci E, Di Maggio G, Petraglia F. Voluminous perinatal pelvic mass: a case of congenital hydrometrocolpos. J Matern Fetal Neonatal Med. 2004;15(2):135-7.

Shaw LM, Jones WA, Brereton RJ. Imperforate hymen and vaginal atresia and their associated anomalies. J R Soc Med. 1983;76(7):560-6.

Heinonen PK. Unicornuate uterus and rudimentary horn. Fertil Steril. 1997;68(2):224-30.

Dane C, Dane B, Erginbas M, Cetin A. Imperforate hymen-a rare cause of abdominal pain: two cases and review of the literature. J Pediatr Adolesc Gynecol. 2007;20(4):245-7.

Nunes N, Karandikar S, Cooper S, Jaganathan R, Irani S. VATER/VACTERL syndrome (vertebra/anus /cardiac/trachea/esophogus/radius/renal/limb anomalies) with a noncommunicating functioning uterine horn and a unicornuate uterus: a case report. Fertil Steril. 2009;91(5):1957.e11-2.

Obeidat RA, Aleshawi AJ, Tashtush NA, Alsarawi H. Unicornuate uterus with a rudimentary non-communicating cavitary horn in association with VACTERL association: case report. BMC Womens Health. 2019;19(1):71.

Roman JD. Co-existence of a rudimentary non-communicating horn with a unicornuate uterus in association with 2 components of the vacterl association: a case report. Case Rep Obstet Gynecol Rep. 2021;3(2):1-4.

Lupean RA, Ștefan PA, Csutak C, Lebovici A, Măluțan AM, Buiga R, et al. Differentiation of Endometriomas from Ovarian Hemorrhagic Cysts at Magnetic Resonance: The Role of Texture Analysis. Medicina (Kaunas). 2020;56(10):487.

Koninckx PR, Barlow D, Kennedy S. Implantation versus infiltration: the Sampson versus the endometriotic disease theory. Gynecol Obstet Invest. 1999;47 Suppl 1:3-9; discussion 9-10.

Matsuura K, Ohtake H, Katabuchi H, Okamura H. Coelomic metaplasia theory of endometriosis: evidence from in vivo studies and an in vitro experimental model. Gynecol Obstet Invest. 1999;47 Suppl 1:18-20; discussion 20-2.

Konrad L, Dietze R, Kudipudi PK, Horné F, Meinhold-Heerlein I. Endometriosis in MRKH cases as a proof for the coelomic metaplasia hypothesis? Reproduction. 2019;158(2):R41-R47.

Abboud K, Giannini A, D'Oria O, Ramadan A, Ayed A, Laganà AS, et al. Laparoscopic Management of Rudimentary Uterine Horns in Patients with Unicornuate Uterus: A Systematic Review. Gynecol Obstet Invest. 2023;88(1):1-10.

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Published

2024-06-27

How to Cite

Bhadwal, A., Sharma, S., & Ranga, S. (2024). Uterine rudimentary horn and ovarian endometriosis with a special emphasis on VACTERL association: a report of two cases. International Journal of Reproduction, Contraception, Obstetrics and Gynecology, 13(7), 1868–1872. https://doi.org/10.18203/2320-1770.ijrcog20241793

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Section

Case Reports