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Herlyn-Werner-Wunderlich syndrome presenting with dysmenorrhea: a case report.
J Med Case Rep 2019; 13(1):323JM

Abstract

BACKGROUND

Herlyn-Werner-Wunderlich syndrome is a rare congenital anomaly characterized by uterus didelphys, obstructed hemivagina, and ipsilateral renal agenesis. The most common presentation is abdominal pain, dysmenorrhea, and abdominal mass secondary to hematocolpos. We present the first case report on Herlyn-Werner-Wunderlich syndrome from Bangladesh.

CASE PRESENTATION

A 15-year-old Asian girl presented with lower abdominal pain of 3 months' duration. She had had menarche 3 months earlier and had a regular menstrual cycle with cyclical abdominal pain. Abdominal examination found a tender mass on the right iliac fossa. Further evaluation with ultrasound revealed distended endometrial cavity filled with complex fluid and nonvisualization of the right kidney. Pelvic magnetic resonance imaging showed absent right kidney and two separate endometrial stripes surrounded by endometrium and a muscular layer. The right endometrial cavity and cervix were distended with blood. This magnetic resonance imaging finding is consistent with Herlyn-Werner-Wunderlich syndrome with uterine didelphyis, right-sided hematometra resulting from obstructed hemivagina, and ipsilateral agenesis of the right kidney. The vaginal septum was resected for vaginoplasty. She was discharged 5 days after surgery and came for follow-up after 7 days. Vaginal examination revealed a healthy wound with no adhesion of the vaginal wall. She also informed us that she had started regular menstruation without any pain 30 days after the operation.

CONCLUSION

An unusual presentation of regular menstruation and nonspecific abdominal pain delays the diagnosis, which can lead to complications such as endometriosis and infertility. Awareness is required; otherwise, misdiagnosis clearly can occur.

Authors+Show Affiliations

Department of Gynaecology and Obstetrics, Cumilla Medical College and Hospital (CuMCH), Cumilla, Bangladesh. sharmennishu29@gmail.com.Department of Radiology and Imaging, Cumilla Medical College and Hospital (CuMCH), Cumilla, Bangladesh.Department of Gynaecology and Obstetrics, Cumilla Medical College and Hospital (CuMCH), Cumilla, Bangladesh.Department of Gynaecology and Obstetrics, Cumilla Medical College and Hospital (CuMCH), Cumilla, Bangladesh.Dhaka Hospital, Nutrition and Clinical Services Division, International Centre for Diarrhoeal Disease Research, Dhaka, Bangladesh.Department of Gynaecology and Obstetrics, Cumilla Medical College and Hospital (CuMCH), Cumilla, Bangladesh.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

31666132

Citation

Nishu, Dilruba Sharmen, et al. "Herlyn-Werner-Wunderlich Syndrome Presenting With Dysmenorrhea: a Case Report." Journal of Medical Case Reports, vol. 13, no. 1, 2019, p. 323.
Nishu DS, Uddin MM, Akter K, et al. Herlyn-Werner-Wunderlich syndrome presenting with dysmenorrhea: a case report. J Med Case Rep. 2019;13(1):323.
Nishu, D. S., Uddin, M. M., Akter, K., Akter, S., Sarmin, M., & Begum, S. (2019). Herlyn-Werner-Wunderlich syndrome presenting with dysmenorrhea: a case report. Journal of Medical Case Reports, 13(1), p. 323. doi:10.1186/s13256-019-2258-6.
Nishu DS, et al. Herlyn-Werner-Wunderlich Syndrome Presenting With Dysmenorrhea: a Case Report. J Med Case Rep. 2019 Oct 31;13(1):323. PubMed PMID: 31666132.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Herlyn-Werner-Wunderlich syndrome presenting with dysmenorrhea: a case report. AU - Nishu,Dilruba Sharmen, AU - Uddin,Md Monir, AU - Akter,Khadija, AU - Akter,Shameema, AU - Sarmin,Monira, AU - Begum,Sartaj, Y1 - 2019/10/31/ PY - 2019/06/17/received PY - 2019/09/10/accepted PY - 2019/11/1/entrez PY - 2019/11/2/pubmed PY - 2019/11/2/medline KW - Herlyn-Werner-Wunderlich syndrome KW - Müllerian anomaly KW - Obstructed hemivagina KW - Vaginoplasty SP - 323 EP - 323 JF - Journal of medical case reports JO - J Med Case Rep VL - 13 IS - 1 N2 - BACKGROUND: Herlyn-Werner-Wunderlich syndrome is a rare congenital anomaly characterized by uterus didelphys, obstructed hemivagina, and ipsilateral renal agenesis. The most common presentation is abdominal pain, dysmenorrhea, and abdominal mass secondary to hematocolpos. We present the first case report on Herlyn-Werner-Wunderlich syndrome from Bangladesh. CASE PRESENTATION: A 15-year-old Asian girl presented with lower abdominal pain of 3 months' duration. She had had menarche 3 months earlier and had a regular menstrual cycle with cyclical abdominal pain. Abdominal examination found a tender mass on the right iliac fossa. Further evaluation with ultrasound revealed distended endometrial cavity filled with complex fluid and nonvisualization of the right kidney. Pelvic magnetic resonance imaging showed absent right kidney and two separate endometrial stripes surrounded by endometrium and a muscular layer. The right endometrial cavity and cervix were distended with blood. This magnetic resonance imaging finding is consistent with Herlyn-Werner-Wunderlich syndrome with uterine didelphyis, right-sided hematometra resulting from obstructed hemivagina, and ipsilateral agenesis of the right kidney. The vaginal septum was resected for vaginoplasty. She was discharged 5 days after surgery and came for follow-up after 7 days. Vaginal examination revealed a healthy wound with no adhesion of the vaginal wall. She also informed us that she had started regular menstruation without any pain 30 days after the operation. CONCLUSION: An unusual presentation of regular menstruation and nonspecific abdominal pain delays the diagnosis, which can lead to complications such as endometriosis and infertility. Awareness is required; otherwise, misdiagnosis clearly can occur. SN - 1752-1947 UR - https://www.unboundmedicine.com/medline/citation/31666132/Herlyn-Werner-Wunderlich_syndrome_presenting_with_dysmenorrhea:_a_case_report L2 - https://jmedicalcasereports.biomedcentral.com/articles/10.1186/s13256-019-2258-6 DB - PRIME DP - Unbound Medicine ER -