Journal List > Korean J Obstet Gynecol > v.54(12) > 1088384

Kim, Park, Ku, Kim, Choi, Kim, and Moon: An unusual presentation of obstructed hemivagina and ipsilateral renal anomaly syndrome: A case report

Abstract

Most patients with syndrome of obstructed hemivagina and ipsilateral renal anomaly (OHVIRA) were presented with dysmenorrhea, pelvic mass and hematocolpos. These common symptoms of OHVIRA syndrome were aggravated soon after the menarche. Early diagnosis is important to relieve the symptom, to prevent injury of genital tract and to preserve fertility. We have experienced an unusual presentation of OHVIRA syndrome, which were presented with vaginal spotting and diagnosed at age 24. Therefore, we present it with a review of the literature.

Figures and Tables

Fig. 1
Transvaginal sonographic findings. (A) Uterine didelphys on coronal view. (B) Hematocolpos on sagittal view.
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Fig. 2
The left upper vagina was dilated and filled with hematoma which was seen as high signal intensity on T2 weighted magnetic resonance imaging. This finding caused by vaginal atresia was consistent with hematocolpos. The annotated tubular structure arising from left upper side of affected vagina was presumed remnant left distal ureter opened to the vagina.
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Fig. 3
The left upper vagina was dilated and filled with hematoma which was seen as high signal intensity on T2 weighted magnetic resonance imaging. This finding caused by vaginal atresia was consistent with hematocolpos. The annotated tubular structure arising from left upper side of affected vagina was presumed remnant left distal ureter opened to the vagina.
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Fig. 4
Post-instillation images of saline infusion sonohysterography. (A) Endometrial cavity of right uterus was dilated normally. (B) But left side was not. There is no evidence of communication between both side of uterine cavity.
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Fig. 5
Laparoscopic finding. There were normal ovaries and tubes arising from each side of uterine cornus. Left lower side of uterine posterior wall was hyperemic and bulged due to hematocolpos. There was a small endometriotic spot on the affected peritoneal surface.
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References

1. Lee BH, Kim JW, Oh SI, Kim MH, Park NH, Lee JY, et al. 3 Cases of uterus didelphys with an obstructed hemivagina and ipsilateral renal agenesis. Korean J Obstet Gynecol. 1997. 40:1489–1495.
2. Pyun JS, Kang H, Choi YW, Jin SJ. A case of uterus didelphys associated with unilateral imperforate vagina and ipsilateral renal agenesis. Korean J Obstet Gynecol. 1981. 24:519–584.
3. Gilliland B, Dyck F. Uterus didelphys associated with unilateral imperforate vagina. Obstet Gynecol. 1976. 48:5S–8S.
4. Smith NA, Laufer MR. Obstructed hemivagina and ipsilateral renal anomaly (OHVIRA) syndrome: management and follow-up. Fertil Steril. 2007. 87:918–922.
5. Zurawin RK, Dietrich JE, Heard MJ, Edwards CL. Didelphic uterus and obstructed hemivagina with renal agenesis: case report and review of the literature. J Pediatr Adolesc Gynecol. 2004. 17:137–141.
6. Shavell VI, Montgomery SE, Johnson SC, Diamond MP, Berman JM. Complete septate uterus, obstructed hemivagina, and ipsilateral renal anomaly: pregnancy course complicated by a rare urogenital anomaly. Arch Gynecol Obstet. 2009. 280:449–452.
7. Gholoum S, Puligandla PS, Hui T, Su W, Quiros E, Laberge JM. Management and outcome of patients with combined vaginal septum, bifid uterus, and ipsilateral renal agenesis (Herlyn-Werner-Wunderlich syndrome). J Pediatr Surg. 2006. 41:987–992.
8. Yang KM, Chae NH, Bae DH, Lim JE, Lee YA, Kang OR, et al. 6 Cases of uterus didelphys associated with obstructed hemivagina and ipsilateral renal agenesis. Korean J Obstet Gynecol. 1998. 41:2022–2026.
9. Hørding U, Legarth J. Uterus didelphys with a unilateral imperforate hemivagina and ipsilateral renal agenesis. Acta Obstet Gynecol Scand. 1987. 66:277–278.
10. Stassart JP, Nagel TC, Prem KA, Phipps WR. Uterus didelphys, obstructed hemivagina, and ipsilateral renal agenesis: the University of Minnesota experience. Fertil Steril. 1992. 57:756–761.
11. Asha B, Manila K. An unusual presentation of uterus didelphys with obstructed hemivagina with ipsilateral renal agenesis. Fertil Steril. 2008. 90:849.e9–849.e10.
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