Human Reproduction, Vol. 14, No. 7, 1741-1743,
July 1999
© 1999 European Society of Human Reproduction and Embryology
Laparoscopic hemi-hysterectomy in treatment of a didelphic uterus with a hypoplastic cervix and obstructed hemivagina: Case report
Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Chang Gung University, Taipei, Taiwan
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Abstract |
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Maldevelopment of the Müllerian duct system may result in various urogenital anomalies including didelphic uterus with a hypoplastic cervix and obstructed hemivagina. We report a patient with this anomaly who was treated by laparoscopic hemi-hysterectomy and hysteroscopic resection of hemivagina. A 17 year old patient who had complained of vaginal pus-like discharge on and off for 1 year was diagnosed by MRI to have a double uterus with obstructed right hemivagina and ipsilateral renal agenesis. After hysteroscopic identification of hypoplasia of the right uterine cervix, laparoscopic resection of the right uterus and right Fallopian tube and hysteroscopically assisted resection of the vaginal septa were performed successfully. From our experience, combined laparoscopy and hysteroscopy may be an efficacious alternative in the management and diagnosis of Müllerian anomalies.
Key words: hemihysterectomy/hysteroscopy/laparoscopy/Müllerian anomaly
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Introduction |
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Didelphic uterus with a hypoplastic cervix and obstructed hemivagina is a rare Müllerian anomaly. It results from abnormal fusion of the Müllerian ducts or from failure of the septum to be absorbed; it may be associated with obstructive phenomena that lead to dysmenorrhoea and abdominopelvic masses. Surgical reconstruction of the internal genitalia, restoration of menses and maintenance of a patent genital tract for these patients are challenging problems for the gynaecologist. Though there is much literature regarding management of these maldevelopments, none so far recommends hysteroscopic and laparoscopic treatment. We report here a case in which the laparoscope and hysteroscope were used in the diagnosis and treatment of didelphic uterus with a hypoplastic cervix and obstructed hemivagina.
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Case report |
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A 17 year old female patient was referred with a 1 year history of dysmenorrhoea and vaginal pus-like discharge. On examination, she was of moderate stature, with normal external genitalia and regular menstruation. Her endocrine profile, including follicle stimulating hormone (FSH), luteinizing hormone (LH), thyroid stimulating hormone (TSH), prolactin and oestradiol, was within normal limits; CA-125 concentration was also within normal limits. The concentrations of ESR and CRP were slightly elevated. The sonogram revealed a condition compatible with didelphic uterus and right haematocolpos. An intravenous pyelography revealed right renal agenesis with normal function and contour of the left renal collecting system. Magnetic resonance imaging suggested a double uterus with double cervix and septate vagina with haematocolpos. Under the impression that the patient had a double uterus with right obstructive hemivagina, she was admitted for operation.
The procedures were performed under general endotracheal anaesthesia through videolaparoscopy. The patient was placed in the dorsolithotomy Trendelenburg position with both legs protected by elastic bandages. A Foley catheter was preoperatively inserted for continuous monitoring of urine output during the operation. The operation began with hymenotomy, and the hysteroscope was then inserted. To provide positive pressure we used a distention pressure of 100–150 mm Hg. To maintain pressure we used a distilled water pump in conjunction with a suction–irrigation unit. Negative pressure was obtained from a suction unit. The outflow port of the resectoscope was connected directly to the suction bottles with suction set on low pressure. Pus-like discharge was noted from a fistular hole on the septate vagina. Obstructed right hemivagina with hypoplasia of the right uterine cervix was noted hysteroscopically (Figure 1
). A normal left uterus and adnexa and normal right ovary were seen without obvious endometriotic lesion, but a right haematosalpinx was noted laparoscopically.
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Pus culture and excision of the vaginal septum were performed first. Later, laparoscopic hemi-hysterectomy was undertaken (Figure 2
). A 10 mm skin incision was made with a scalpel at the umbilicus, and this site was used as the point of access for both the Veress needle and the trocar (Figure 3). The Veress needle (Auto-Suture, Norwalk, CT, USA) was then inserted perpendicular to the abdominal wall. After adequate pneumoperitoneum was obtained, a 10 mm trocar and the laparoscope were inserted, and two more 5 mm puncture sites were made in the lower abdomen at the para-median line, just above the pubic hair line; one 12 mm puncture site was then made at the para-median line at the level of the umbilicus. A total of four trocars was required (Figure 3).
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The operation began with the coagulation and dissection of the right round ligaments. The anterior leaves of the broad ligaments were opened and incised with laparoscopic scissors to the point of the vesico-uterine pouch. The parametrium was then dissected along the uterus after coagulation. The pouch was opened by means of laparoscopic scissors. The uterine artery was dissected with Kleppinger bipolar forceps and then divided with scissors. The remaining right uterine tissue was separated by an automatic endoscopic stapler and the freed right uterus was removed vaginally through posterior colpotomy. The myometrium was approximated by 1-0 Prolene (Ethicon Inc., Somerville, NJ, USA) sutures. The entire pelvic cavity was inspected and irrigated with lactated Ringer's solution. All debris and blood were removed. The 10 mm and 12 mm incision wounds along the abdomen were closed with Mattress sutures; the other puncture sites were, meanwhile, approximated by sterile adhesive tape (Lee and Soong, 1993
). The post-operative period was uneventful, and the patient was discharged 5 days later. The main operative findings can be summarized as follows: (i) grossly normal left uterus and adnexa; (ii) normal right uterus with hypoplasia of the cervix with right haematosalpinx; (iii) normal right ovary; (iv) longitudinal septa resulting in an obstructed right hemivagina with small fistulae; (v) no obvious endometriosis. Laparoscopic resection of right uterus, cervix and right haematosalpinx and resection of vaginal septa were performed smoothly. The pathology reports revealed vaginal mucosa, the endometrium and cervix of the uterus having chronic inflammation. Eschericia coli was isolated from the vaginal purulent discharge. The patient continues to have regular menses without dysmenorrhoea, now 2 years postoperatively.
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Discussion |
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TopAbstractIntroductionCase reportDiscussionReferences |
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Maldevelopment of the Müllerian ducts may result in various urogenital anomalies. The association between didelphic uterus with a hypoplastic cervix and obstructed hemivagina is relatively uncommon and difficult to detect (Magee et al., 1979
). It may result in a unilateral abdominopelvic mass terminating in a purpuric bulge of the lateral vaginal wall in a young woman with progressively severe dysmenorrhoea (Rock et al., 1980). Ideally, the anomaly should be completely defined before the operation. Often, however, the extent of the anomaly can only be properly determined at surgery (Pinsonneault and Goldstein, 1985). In our experience, the usage of laparoscopy combined with operative hysteroscopy is helpful in management and accurate categorization of these anomalies (Lee et al., 1999).
The treatment of patients with a combination of vaginal and uterine obstruction is determined by the presence of the cervix (Stangl et al., 1983; Fried et al., 1987). Because of postoperative complications of uterovaginal canalization, including peritonitis and recurrent obstruction of the uterovaginal neocanal, the majority of clinicians view hysterectomy as the optimal primary surgical management in these patients (Victor et al., 1997). Haematosalpinx, haematocolpos and hypoplasia of the cervix were present on the right side in our patient. Therefore, right uterus and Fallopian tube resection was performed.
The use of operative laparoscopy combined with hysteroscopy as an efficacious technique in the treatment of reproductive and gynaecological lesions is well known (Lee et al., 1995). Laparoscopic hemihysterectomy is a rather sophisticated technique. However, the automatic endoscopic stapler and suturing technique make these procedures possible. Thus, endoscopy may be a simple and effective alternative in treating certain Müllerian anomalies.
In summary, maldevelopment of the Müllerian ducts may result in various urogenital anomalies. Early and accurate diagnosis of these various types of Müllerian duct anomalies is important. It may avoid unnecessary hysterectomy, and fertility can be preserved in some kinds of Müllerian anomalies. In our experience, laparoscopy and hysteroscopy are useful not only in categorizing these anomalies but also in managing certain Müllerian anomalies. Laparoscopic hemihysterectomy may be a simple and effective alternative in treating a patient with didelphic uterus with a hypoplastic cervix.
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Notes |
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1 To whom correspondence should be addressed at: Department of Obstetrics and Gynecology, Chang Gung Memorial Hospital, Linkou Medical Center, 5, Fu-Hsin Street, Kwei-Shan, Tao-Yuan, Taiwan
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References |
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Fried, A.M., Cliff, M., Wilson, E.A. and Whisnant, J. (1987) Uterine anomalies associated with renal agenesis: role of Gray scale ultrasonography. Am. J. Roentgenol., 131, 973.
Lee, C.L. and Soong, Y.K. (1993) Laparoscopic hysterectomy with the Endo GIATM30. J. Reprod. Med., 15, 582–586.
Lee, C.L., Lai, Y.M. and Soong, Y.K. (1995) Laparoscopic ovariopexy. Hum. Reprod., 10, 372–374.
Lee, C.L., Wang, C.J., Liu, Y.H. et al. (1999) Laparoscopically assisted full thickness skin graft for reconstruction in congenital agenesis of vagina and uterine cervix. Hum. Reprod., 14, 928–930.
Magee, M.C., Lucey, D.T. and Fried, F.A. (1979) A new embryonic classification for urogynecologic malformation: the syndromes of mesonephric duct induced Müllerian deformities. J. Urol., 121, 265.[Web of Science][Medline]
Pinsonneault, O. and Goldstein, D.P. (1985) Obstructing malformations of the uterus and vagina. Fertil. Steril., 44, 241–247.[Web of Science][Medline]
Rock, J.A. and Jones, H.W. Jr (1980) The double uterus associated with an obstructed hemivagina and ipsilateral renal agenesis. Am. J. Obstet. Gynecol., 138, 339–342.[Web of Science][Medline]
Stangl, W., Frank, R.C., Frank, W. et al. (1983) Sonographic findings in a case of uterine and vaginal duplication (didelphys) with unilateral hematocolpometrasalpinx. J. Clin. Ultrasound., 11, 40–41.[Web of Science][Medline]
Victor, Y.F., Heath, M., Nancy, A.K. and Michael, R.S. (1997) Congenital cervical atresia: report of seven cases and review of the literature. Am. J. Obstet. Gynecol., 177, 1419–1425.[Web of Science][Medline]
Submitted on December 7, 1998; accepted on March 11, 1999.
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