Herlyn-werner-wunderlich syndrome: MRI findings, radiological guide (two cases and literature review), and differential diagnosis

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• 作者：Riccardo Del Vescovo (1)
  Sofia Battisti (1)
  Valerio Di Paola (1)
  Claudia L Piccolo (1)
  Roberto L Cazzato (1)
  Ilaria Sansoni (1)
  Rosario F Grasso (1)
  Bruno Beomonte Zobel (1)
  
• 刊名：BMC Medical Imaging
• 出版年：2012
• 出版时间：December 2012
• 年：2012
• 卷：12
• 期：1
• 全文大小：354KB
• 参考文献：1. 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 (Herlin-Werner-Wunderlich syndrome. / J Pediatr Surg 2006,41(5):987-2. CrossRef
  2. Alan JW, Louis RK: / Campbell-Walsh Urology. 9th edition. Philadelphia: Saunders; 3270-.
  3. Orazi C, Lucchetti MC, Schingo PM, Marchetti P, Ferro F: Herlyn-Werner-Wunderlich syndrome: uterus didelphys, blind hemivagina and ipsilateral renal agenesis. Sonographic and MR findings in 11 cases. / Pediatr Radiol 2007, 37:657-5. CrossRef
  4. Lee BH, Kim JW, Oh SI, / et al.: 3 cases of uterus didelphys with obstructed hemivagina and ipsilateral renal agenesis. / Korean J Obstet Gynecol 1997, 40:1489-5.
  5. Heinonen PK: Clinical implications of the didelphic uterus: long-term follow-up of 49 cases. / Eur J Obstet Gynecol Reprod Biol 2000, 91:183-0. CrossRef
  6. Candiani GB, Fedele L, Candiani M: Double uterus, blind hemivagina and ipsilateral renal agenesis: 36 cases and long-term follow-up. / Obstet Gynecol 1997, 90:26-2. CrossRef
  7. Park Noh Hyuck, Park Hee Jin, Park Chan Sup, Park Sung Il: Herlyn-Werner-Wunderlich Syndrome with Unilateral Hemivaginal Obstruction, Ipsilateral Renal Agenesis and Contralateral Renal Thin GBM Disease: A Case Report with Radiological Follow Up. / J Korean Soc Radiol 2010, 62:383-88.
  8. 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, 17:137-1. CrossRef
  9. Kim TE, / et al.: Hysteroscopic resection of the vaginal septum in uterus didelphys with obstructed hemivagina: a case report. / J Korean Med Sci 2007, 22:766-69. CrossRef
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  14. Foglia RP, Kim SH, Cleveland RH, Donahoe PK: Complications of vaginal atresia in association with a duplicated Müllerian duct. / J Pediatr Surg 1987,22(7):653-56. CrossRef
  15. Haddad B, Barranger E, Paniel BJ: Blind hemivagina: long-term follow-up and reproductive performance in 42 cases. / Hum Reprod 1999,14(8):1962-964. CrossRef
  16. Kiechl-Kohlendorfer U, Geley TE, Unsinn KM, Ganer I: Diagnosing neonatal female genital anomalies using saline-enhanced sonography. / AJR Am J Roentgenol 2001, 177:1041-044.
  17. Burgis J: Obstructive Müllerian anomalies: Case report, diagnosis, and management. / Am J Obstet Gynecol 2001, 185:338-4. CrossRef
  18. Gruenwald P: Relation of the growing Müllerian duct to the Wolffian duct and its importance for the genesis of malformation. / Anat Rec 1941, 81:1-0. CrossRef
  19. Park Noh Hyuck, Park Hee Jin, Park Chan Sup, Park Sung Il: Herlyn-Werner-Wunderlich Syndrome with Unilateral Hemivaginal Obstruction, Ipsilateral Renal Agenesis and Contralateral Renal Thin GBM Disease: A Case Report with Radiological Follow Up. / J Korean Soc Radiol 2010, 62:383-88.
  20. Brosens IA, Brosens JJ: Endometriosis. / Eur J Obstet Gynecol Reprod Biol 2000,90(2):159-4. CrossRef
  21. 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, 280:449-2. CrossRef
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  23. The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2342/12/4/prepub
  
• 作者单位：Riccardo Del Vescovo (1)
  Sofia Battisti (1)
  Valerio Di Paola (1)
  Claudia L Piccolo (1)
  Roberto L Cazzato (1)
  Ilaria Sansoni (1)
  Rosario F Grasso (1)
  Bruno Beomonte Zobel (1)
  
  1. Department of Radiology, Campus Bio-Medico Univeristy of Rome, Rome, Italy
  

文摘

Background Herlyn-Werner-Wunderlich (HWW) syndrome is a very rare congenital anomaly of the urogenital tract involving Müllerian ducts and Wolffian structures, and it is characterized by the triad of didelphys uterus, obstructed hemivagina and ipsilateral renal agenesis. It generally occurs at puberty and exhibits non-specific and variable symptoms with acute or pelvic pain shortly following menarche, causing a delay in the diagnosis. Moreover, the diagnosis is complicated by the infrequency of this syndrome, because Müllerian duct anomalies (MDA) are infrequently encountered in a routine clinical setting. Cases presentation two cases of HWW syndrome in adolescents and a differential diagnosis for one case of a different MDA, and the impact of magnetic resonance (MR) imaging technology to achieve the correct diagnosis. Conclusions MR imaging is a very suitable diagnostic tool in order to perform the correct diagnosis of HWW syndrome.