Blind hemivagina: long-term follow-up and reproductive performance in 42 cases

B. Haddad1, E. Barranger and B.J. Paniel

Service de Gynécologie-Obstétrique, CHI Créteil, 40 avenue de Verdun, 94010 Créteil, France


    Abstract
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 Abstract
 Introduction
 Materials and methods
 Results
 Discussion
 References
 
Our purpose was to analyse the reproductive performance of women with obstructed hemivagina after surgical treatment. After laparoscopic exploration of 42 cases (mean age: 18 years), didelphic and complete septate uterus were found in 78 and 22% of cases respectively. Resection of vaginal septum and hemihysterectomy with ipsilateral hemicolpectomy were performed in 88% and 12% of the cases, respectively, between 1970 and 1997. Long-term results were assessed by a questionnaire and obtained for 38 patients (mean years after treatment and range: 6.5; 1–23). Dysmenorrhoea and abdominal pain were resolved in 87% and 100% of the cases, respectively. Nine patients experienced 20 pregnancies (13 living children, four early spontaneous abortions, two early terminations and one ectopic pregnancy). Nine offspring (69% of live births) were delivered after 37 weeks. Four patients had four pregnancies ipsilateral to blind hemivagina after vaginal septum resection (two living children, one early spontaneous abortion and one ectopic pregnancy). These results suggest that laparoscopic exploration and resection of vaginal septum are the appropriate treatments for obstructed hemivagina. Subsequent reproductive performance was comparable to that reported following treatment of the associated uterine malformation.

Key words: blind hemivagina/pregnancy outcome/renal agenesis/uterine malformation


    Introduction
 Top
 Abstract
 Introduction
 Materials and methods
 Results
 Discussion
 References
 
Blind hemivagina is a rare malformation which involves Müllerian and Wolffian ducts. Although the condition was first recognized in 1922 (Purslow, 1922Go) and is represented by the presence of a didelphic uterus and ipsilateral renal agenesis, the pathogenesis remains unclear and its aetiology is still unknown. Most reports concerned small series, the maximum being a series of 36 patients (Candiani et al., 1997Go). Conservative surgical treatment (excision of the obstructing septum followed by a marsupialization of the blind hemivagina) is generally regarded as appropriate. However, long-term functional results and reproductive performance after surgical treatment have not been well established. We therefore studied a series of 42 cases in order to evaluate these parameters.


    Materials and methods
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 Abstract
 Introduction
 Materials and methods
 Results
 Discussion
 References
 
We carried out a retrospective study of patients with a blind hemivagina who were treated in our centre between 1970 and July 1997. A total of 42 patients (mean age: 18 years, range: 11–30) were reviewed. four women had five pregnancies before referral, two of whom (didelphic and complete septate uterus) had three pregnancies. These latter pregnancies took place in the ipsilateral uterus and were delivered before 37 weeks by Caesarean section; a communication between blind and normal vagina was found in these two patients. Two other patients (didelphic and complete septate uterus) had two pregnancies which occurred in the normal cavity and were terminated early. These five pretreatment pregnancies were not included in the results.

Symptoms at time of referral are outlined in Table IGo. Right blind hemivagina was found in 52% of patients. All patients underwent ultrasound examination of the kidneys or intravenous pyelography: an ipsilateral renal agenesis was found in all cases except for one patient who had normal kidneys and a didelphic uterus.


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Table I. Patient characteristics (n = 42)
 
Pelvic ultrasonography and, where possible, hysterosalpingography were performed in 74 and 19% of the cases, respectively. All patients except one underwent laparoscopy prior to the surgical procedure, in order to clarify the uterine malformation (as a function of American Fertility Society Classification, 1988) and to explore the upper genital tract. The exception underwent resection of vaginal septum in 1970 without prior laparoscopy; 18 years later this patient underwent laparoscopy for Fallopian tube ligation. Although this permitted exploration of the genital tract, the results were excuded from this study. Didelphic and complete septate uterus were found in 78 and 22% of cases, respectively. Associated complications of the upper genital tract were found in 39% of cases (Table IIGo). Endometriosis, when present, was stage I, II, III in 11, one and three cases, respectively (revised American Fertility Society Classification).


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Table II. Upper genital tract complications (n = 41)
 
Surgical treatment consisted of vaginal septum excision in 37 patients (88%). Vaginal septum excision was performed in one procedure for 34 patients. Two interventions were needed for three patients (one with a pyocolpos and two with a haematocolpos which reached the hymeneal ring): a limited resection–marsupialization (nearly 3 cm diameter) and drainage of the blind hemivagina was followed 1 month later by the resection of the septum. Associated complications of the upper genital tract were treated after resection of the septum (Table IIIGo).


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Table III. Associated surgery performed for upper genital tract complications in women undergoing resection of the septum (n = 37)
 
Hemihysterectomy and ipsilateral hemicolpectomy were performed in five patients (12%) with severe adnexal complications including adhesions between Fallopian tubes and ovaries and Fallopian tube lacerations. Two of these patients also had an associated oophorectomy. Two severe complications arose in these patients. In one case blood transfusion was needed after the surgical intervention and in the second the presence of a pelvic haematoma required a second intervention 14 days later. All patients received broad-spectrum antibiotics (Ampicillin) during surgical treatments.

Patients were examined 3 months later. Morphological results were obtained from medical records. To assess long-term reproductive performance the patients were asked by phone to complete a questionnaire; this occurred an average of 6.5 years after surgical procedure (range: 1–23 years). The questionnaire concerned dysmenorrhoea, vaginal discharge, dyspareunia and pregnancies, including their number and results (live birth, early termination, early spontaneous abortion and ectopic pregnancy), gestational age at delivery, mode of delivery and whether the pregnancy was ipsi- or contralateral to the obstructed hemivagina.


    Results
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 Abstract
 Introduction
 Materials and methods
 Results
 Discussion
 References
 
Surgical treatment assessed after 3 months appeared to be satisfactory in 39 cases (93%). In three cases (7%), a minor and ipsilateral stricture was observed at the lateral side of the vagina, near the fornix, twice after hemihysterectomy and ipsilateral hemicolpectomy and once after conservative treatment. Further surgery was not required.

Thirty-eight patients (90%) answered the questionnaire. Dysmenorrhoea and abdominal pain were resolved in 87% (27/31) and 100% of cases, respectively. Vaginal discharge was noted in 16% of cases. Dyspareunia was resolved in both patients (Table IGo), and no new cases were noted after surgery.

With regard to reproductive performance, eight patients had not attempted intercourse, 19 did not wish to become pregnant, two had been trying for 6 and 12 months respectively to achieve pregnancy. Nine women who had undergone vaginal septum excision experienced 20 pregnancies after surgical procedure and the results are outlined in Table IVGo. Four women had 4 ipsilateral pregnancies after resection of vaginal septum. Two of them (with a didelphic and complete septate uterus) had vaginal deliveries after 37 weeks and two others (with didelphic uterus) had an early spontaneous abortion and an ectopic pregnancy. The five patients who underwent hemihysterectomy and ipsilateral hemicolpectomy did not achieve pregnancy.


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Table IV. Reproductive performance of women with blind hemivagina after surgical procedure (n = 9)
 

    Discussion
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 Abstract
 Introduction
 Materials and methods
 Results
 Discussion
 References
 
Uterine malformation, when associated with obstructed hemivagina and ipsilateral renal agenesis, has been generally described as a double, mainly didelphus, uterus (Rock and Jones, 1980Go; Morgan et al., 1987Go; Stassart et al., 1992Go; Candiani et al., 1997Go). However, the external shape of uterus was frequently unknown since diagnosis was generally achieved by hysterosalpingography. Rare cases of septate uterus have been reported (Vinstein and Franken, 1972Go; Robert and Le Charpentier, 1974Go; Yoder and Pfister, 1976Go; Rock and Jones, 1980Go; Chelli et al., 1994Go). In our study, laparoscopic exploration showed a complete septate uterus in nine cases (22%).

Ipsilateral renal agenesis is prevalent since the development of the urinary system parallels that of the genital tract (Muller et al., 1967Go; Acien, 1992Go). However, the presence of normal kidneys has been reported (Johnson and Hillman, 1986Go) and we found a case of a normal urinary tract in our series. It has been reported that the right one is predominantly involved, occurring in 66% of cases (Rock and Jones, 1980Go; Morgan et al., 1987Go). However, our results did not confirm this, right and left sides being equally involved.

Clinical management of blind hemivagina must include renal imaging by ultrasonography or intravenous pyelography, both to confirm the absence of a normal kidney in the affected side and to detect abnormalities in the contralateral urinary tract (Stassart et al., 1992Go). Our series included a case of contralateral vesico-ureteral reflux with a normal kidney which necessitated surgical treatment. Assessment of uterine malformation by hysterosalpingography may not be beneficial (Acien, 1997Go), though it may permit a communicating uterus to be diagnosed especially in patients with haematic vaginal discharge. Abdominal and endovaginal ultrasonography (Nasri et al., 1990Go) and more recently three-dimensional ultrasound (Jurkovic et al., 1995Go; Raga et al., 1996Go) may contribute to the analysis of the external uterine shape. Magnetic resonance imaging may also be helpful to detect uterine anomalies (Mintz et al., 1987Go; Pellerito et al., 1992Go; Sardanelli et al., 1995Go). However, these two latter techniques are expensive and do not change radically the management of this malformation. Laparoscopic exploration has the ability to assess the type of uterine malformation and reveal other complications in the upper genital tract which may require appropriate surgery. Early correct diagnosis will allow the appropriate surgical treatment to be performed in a single procedure, including laparoscopic exploration and resection of the vaginal septum. Incision of the septum followed by resection 2–3 months later (Morgan et al., 1987Go) should be avoided as incision alone may lead to the development of haematocolpos or pyocolpos after a spontaneous closure. However, to avoid resection of normal vaginal tissue, particularly when the obstructed hemivagina reaches the hymeneal ring, a limited resection–marsupialization (3 cm diameter) may be performed during an initial surgical procedure, allowing the remaining vaginal septum to be removed 1 month later. This was performed three times in this study, twice for haematocolpos and once for pyoclopos. In the second case of pycolpos, resection of vaginal septum was performed in one surgical procedure and was not affected by infected menstrual retention (Candiani et al., 1997Go).

The obstetric outcome in our series was similar to other studies (Acien, 1993Go; Raga, 1997Go). Pregnancies occurred mainly in the contralateral cavity (80%), although four patients had pregnancies in the affected side after surgical procedure. These latter findings confirm that, once the obstruction has been relieved, both the dilated uterus and its corresponding tube recover their normal function.

In conclusion, our results confirm that diagnosis of blind hemivagina should be made as early as possible to avoid genital complications which may necessitate aggressive surgical treatment. Resection of the vaginal septum preceded by laparoscopic exploration of the upper genital tract allows uterine anatomy and function to recover. Obstetric outcome after surgical treatment depends on the type of uterine malformation present.


    Notes
 
1 To whom correspondence should be addressed, c/o Baha M.Sibai, MD, Division of Maternal–Fetal Medicine, University of Tennessee, 853 Jefferson Avenue, Memphis, TN 38163, USA Back


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Submitted on December 30, 1998; accepted on April 9, 1999.