Minimally Invasive Therapy.

2010;19:241247

CASE REPORT

Diagnosis and laparoscopic management of a rudimentary uterine horn in a teenage girl, presenting with haematometra and severe endometriosis: Our experience and review of literature

SPIROS A. LIATSIKOS1, PANAGIOTIS TSIKOURAS1, VASILEIOS SOUFTAS2, ALEXANDROS AMMARI1, PANAGIOTIS PRASSOPOULOS2, GEORGIOS MAROULIS1, VASILEIOS LIBERIS1
1

Department of Obstetrics and Gynecology, Democritus University of Thrace, Greece, and 2Department of Radiology, Democritus University of Thrace, Greece

Summary A unicornuate uterus is a rare congenital malformation of the female genital tract, which appears in about 1/1000 women and is characterized by signicant anatomic variability. In the most common type, a noncommunicating rudimentary horn coexists with the unicornuate uterus. The diagnosis of this anomaly is usually delayed, as it remains asymptomatic until adolescence and its initial symptoms are atypical. As a result, it is often diagnosed after the appearance of severe complications, such as haematometra, endometriosis, infertility and ectopic pregnancy. We report a case of a teenage girl presenting with dysmenorrhoea, endometriosis and haematometra secondary to a noncommunicating rudimentary horn. The diagnosis of the anomaly was based on magnetic resonance imaging (MRI) and laparoscopy. The excision of the symptomatic rudimentary horn and the ipsilateral fallopian tube was also performed laparoscopically. A review of the literature follows, focusing mainly on the diagnosis and laparoscopic management of a unicornuate uterus and its complications in adolescence. Laparoscopy is an accurate diagnostic tool, which also carries signicant advantages in effective surgical management of congenital uterine anomalies, especially in young women.

Key words: Adolescence, unicornuate uterus, endometriosis, laparoscopy

Introduction Congenital malformations of the female genital tract are mainly the result of four types of disturbance during fetal life, concerning the development, formation or fusion of the Mllerian ducts:
. . . .

Failure of one or both ducts to develop (agenesis; unicornuate uterus without rudimentary horn); failure of the ducts to canalize (unicornuate uterus with rudimentary horn, without proper cavities); failure of, or abnormal fusion of, the ducts (uterus didelphys; bicornuate uterus); and failure of reabsorption of the midline uterine septum (septate uterus; arcuate uterus) (14).

The entirely separate origin of the ovaries from the gonadal ridges results in the infrequent association of the genital tract anomalies with ovarian anomalies. Based on the previous work by Buttram (5), the American Society of Reproductive Medicine (ASRM) (1) classied the anomalies of the female genital tract into six major uterine anatomic types, according to the degree of failure of normal development: (I) hypoplasia/agenesis; (II) unicornuate uterus (IIa: with a communicating rudimentary horn; IIb: with a non-communicating rudimentary horn; IIc: with a rudimentary horn without a cavity; IId: without a rudimentary horn); (III) didelphys uterus; (IV) bicornuate uterus (IVa: complete; IVb: partial); (V) septate uterus (Va: complete; Vb partial); (VI) arcuate uterus; (VII) diethylstilbestrol (DES) exposed uterus (T-shaped uterus).

Correspondence: P. Tsikouras, Lysimachou/ Petrina, 68100 Alexandroupolis Greece. E-mail: ptsikour@med.duth.gr ISSN 1364-5706 print/ISSN 1365-2931 online 2010 Informa UK Ltd DOI: 10.3109/13645701003644491

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S. A. Liatsikos et al. CA-153 were also within the normal range, but CA-125 was slightly raised (37). Transabdominal ultrasound revealed a uterus with an abnormal morphology and two homogenous hypoechoic masses lying on the left side of the pelvis, one bigger and globular and the other smaller and longitudinal. Both ovaries were normal. Finally, a quantity of uid was evident in the Douglas pouch. An abdominopelvic magnetic resonance imaging (MRI) conrmed a congenital anomaly of the uterus (Figure 1). A unicornuate uterus was seen projecting to the right side of the pelvis. A rudimentary left horn was also observed, with a cavity that did not communicate with the unicornuate uterus or the vagina (type IIb anomaly according to ASRM classication) (Figure 2). The rudimentary left horn appeared with a myometrium of normal thickness, normal magnetic signal and a discernible broad junctional zone, but with a dilated endometrial cavity containing a substance with a magnetic signal indicative of blood (methemoglobin). A serpentine tubular structure was also observed just adjacent to the rudimentary horn, containing a substance with the same characteristics, probably corresponding to a haematosalpinx. Both ovaries were recognised as normal. Finally, both kidneys were recognised at their typical position and there was no obvious anomaly of the urinary tract. The patient underwent diagnostic and operative laparoscopy. The ndings included a developed right unicornuate uterus with a patent fallopian tube, a normal right ovary and a cervix. A Mllerian remnant

Unicornuate uterus covers a wide range of anatomic variability. The classication of this uterine anomaly according to ASRM depends on the status of a rudimentary horn. It includes a communicating (with the functioning unicornuate uterus) and a noncommunicating rudimentary horn, a horn with or without a cavity and an absent rudimentary horn. All these cases are accompanied by an abnormal external uterine morphology. A brous or brous muscular band sometimes connects the main and the rudimentary horn, but in 89-90% of cases there is no communicating channel between the two uterine cavities. The unicornuate uterus with a noncommunicating rudimentary horn and no cavity seems to be the most common type of this anomaly (~48%) (6,7). Here, we report a case of laparoscopic management of a unicornuate uterus with noncommunicating functional horn, complicated with haematometra, haematosalpinx and severe peritoneal endometriosis (stage IV, according to ASRM staging system), in a teenage girl presenting with dysmenorrhoea and acute abdominal pain.

Case report A 15-year-old virgin woman presented to our emergency department with severe abdominal pain, located mainly in the left lower quadrant, and nausea. An intermittent left lower abdominal pain was noted for the rst time about a month ago, but it became persistent by the day she came to the hospital. Progressive dysmenorrhoea since menarche, at the age of 13, was also mentioned, but her symptoms worsened dramatically over the last six months and were not relieved after medical management (non-steroidal anti-inammatory drugs (NSAIDs)). The patient had a normal pubertal development, but she reported irregular menstruation, mostly a cycle of 40-50 days. There was no relevant past general medical history or previous uterine or pelvic surgery. She and her mother did not have a history of diethylstilbestrol (DES) exposure. Physical examination revealed normal secondary sexual characteristics. The appearance of her external genitalia was unremarkable, with normal pubic hair and an intact hymen. Her physical condition was good. However, abdominal palpation revealed a slight tenderness of the lower abdomen. Her leukocyte count was 10500/cm3. The general serum and urinary biochemical control was normal. Baseline endocrine tests were within the normal range and in accordance with the time of the cycle. Serum b-human chorionic gonadotrophin (b-HCG) titer was negative (< 5 mIU/ml). The tumour indeces CA-199 and

Figure 1. MRI of the congenital anomaly of the uterus. A rudimentary left horn with a cavity did not communicate with the unicornuate uterus or the vagina (type IIb anomaly according to ASRM classication).

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Figure 2. The rudimentary left horn appeared with a myometrium of normal thickness and normal magnetic signal, but with a dilated endometrial cavity.

was seen on the left side of the pelvis, which represented a rudimentary horn. This remnant was attached to the uterus by a small brous band, but did not communicate with the uterine cavity or the cervix. The rudimentary horn was dilated by a chocolate-like content, comprising a haematometra. A sizeable left haematosalpinx was also noted. Severe endometriosis was obvious, involving the left ovary, the left haematosalpinx, the peritoneum of the Douglas pouch and the left uterosacral ligaments. Fibrous adhesions involving the left ovary, the left haematosalpinx, the intestine and the left pelvic sidewall were described. Lysis of left adnexa adhesions was then decided, followed by removal of the haematosalpinx. The haematometra was drained and the procedure was terminated to allow an efcient preparation prior to denite surgery. Following six months of gonadotrophin-releasing hormone (GnRH) analogue therapy, the patient underwent a second operative laparoscopy. The rudimentary horn was freed from adjacent structures (including the unicornuate uterus) and removed. Finally, electrocauterization of remaining endometriosis spots on the pelvic sidewall and the Douglas pouch was performed. The patients hospital course was uneventful after both surgeries and she was discharged on the second postoperative day. Histology was consistent with a noncommunicating rudimentary horn with a functional endometrium, haematometra and haematosalpinx. Six months after the denite surgery, the patient reported regular menstrual cycles, without dysmenorrhoea. Discussion The normal development of the female genital tract requires the persistence and differentiation of the

Mllerian or paramesonephric ducts, which have been formed by an invagination of the coelomic epithelium. Mllerian and Wolfan ducts represent the primitive structures of the internal reproductive organs of females and males respectively. In the beginning, both ducts exist side by side in the human embryo, until genetic sex conducts the differentiation of either ovaries or testes. In genetic males (46,XY), the newly formed testes produce testosterone and Anti-Mullerian Hormone (AMH), which cause the regression of the Mllerian ducts. In genetic female embryos (46,XX), due to the absence of testes and thus AMH, the Mllerian ducts are conserved and form the fallopian tubes, the uterus, the cervix and the upper part of the vagina. On the other hand, the Wolfan ducts regress as a result of the absence of testosterone (810). Unicornuate uterus is the result of complete or partial lack of development or canalization of one of the Mllerian ducts during the early stages of embryogenesis (weeks 7-8 of gestation). The prevalence of this type of uterine malformation has been reported to range between 2,4% and 13,7% of all cases of uterine anomalies (11). However, the mean incidence as mentioned above is estimated to be ~9,6% (12). More interesting is the prevalence of unicornuate uterus in the general polulation. In the review of selected series by Saravelos et al. (13), the prevalence of unicornuate uterus in the general/fertile polulation ranges between 1 in 4000 women and 1 in 1000 women, depending on the diagnostic tool used. Keeping in mind that most studies, which were closer to the incidence of 1:1000 women (1416), used hysterosalpingography with laparoscopy (a relatively accurate method for diagnosing unicornuate uterus) the rate of 1 in 1000 seems to be closer to the true prevalence. Although a unicornuate uterus with a rudimentary horn is rare, it is susceptible to a number of gynaecologic and obstetric complications, such as endometriosis, primary infertility, haematometra and urinary tract anomalies (1719). A unicornuate uterus has been associated with an impaired reproductive outcome. Finally, rupture of a pregnant rudimentary horn is a well known severe complication of the particular anomaly. A noncommunicating cavitated rudimentary horn with a functioning endometrium causes cryptomenorrhoea, which leads to haematometra, dysmenorrhoea and lower abdominal pain soon after menarche, due to hormonal stimulation (20). A haematometra is rarely diagnosed in teenage girls and only after clinical manifestation (dysmenorrhoea, acute pelvic pain, pelvic mass). In that time it is mainly caused by an obstructive uterine anomaly, such as a functional, noncommunicating rudimentary horn.

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S. A. Liatsikos et al. complications. Very rarely though, the pregnancy occurs in the rudimentary horn, probably due to a transperitoneal migration of spermatozoa (25). The incidence of pregnancy in the malformed rudimentary horn is between 1/140000 and 1/100000 (26,27). This condition has been associated with a 70% risk for rudimentary horn rupture occurring between 10 and 15 weeks of gestation (26), as the muscle of the rudimentary horn is exceptionally thin, with a poor capability of distension, and in most cases the placentation is pathological (23). It is always an emergency case, as it is accompanied by massive and life threatening intraperitoneal bleeding. Thus, if pregnancy in the rudimentary horn is diagnosed, the excision of the horn is necessary, although sporadic cases of a living infant have been reported (28). There is a close relationship between the fetal development of the female genital tract and the urinary tract. Initially the Mllerian and Wolfan ducts exist side by side in the human embryo. Damage to either duct may be associated with damage to both. Thus, Mllerian duct anomalies are usually associated with renal abnormalities. According to some studies (29,30) the presence of unilateral renal agenesis will predict an ipsilateral Mllerian anomaly in more than half of the cases. Woolf and Allen (17) reported that unilateral renal agenesis is mostly associated with unicornuate uterus. The reported incidence of major renal anomalies associated with a unicornuate uterus with a noncommunicating rudimentary horn ranges from 31 to 100% (5,31). The most common anomaly is renal agenesis ipsilateral to the rudimentary horn, while the second most commonly reported anomaly is an ipsilateral pelvic kidney (31). It is also reported that unilateral renal agenesis is associated with pregnancy-induced hypertension (7). Therefore, whenever a uterine anomaly is found, an evaluation of the renal system is indicated, using ultrasonography or MRI. Intravenous pyelography offers a more accurate and outright imaging of the urinary tract. In our case, both kidneys were recognized as normal on MRI and no abnormality of the urinary system was detected. A unicornuate uterus with a rudimentary horn is not easily diagnosed before the occurrence of clinical symptoms, especially in a young virgin woman with normal external genitalia. Most cases of such an anomaly are usually found unexpectedly later in the womans life, during infertility evaluation or in clinical manifestation. Of the forty-two women with unicornuate uterus reported by Heinonen (7) in a large retrospective study, only two had a suggestion of this uterine anomaly before the diagnostic procedure. Furthermore, a complete physical examination, which includes a bimanual examination, is not possible in a

Nezhat and Smith (20) reported an interesting case of haematometra in an 18-year-old woman with a unicornuate uterus and two cavitated, noncommunicating rudimentary horns. Lin et al. (21) reported a case of acute abdomen caused by haematometra and haematosalpinx four months following right oophorectomy in a teenage girl with a rudimentary noncommunicating uterine horn. The prevalence of haematometra caused by a noncommunicating rudimentary horn is an issue of controversy. According to a histopathologic study by Fedele et al. (22), there is rarely a functional endometrium in a cavitary rudimentary horn and therefore a haematometra is not a common complication of these women. According to Heinonen (18), haematometra was found in only one out of 15 patients with a noncommunicating rudimentary horn with a cavity. However the prevalence of haematometra is signicantly higher in the study of Liu (23): Of the 22 cases of unicornuate uterus with a rudimentary horn reported, haematometra occurred in ve (22,7%). The prevalence is even higher in adolescence: Haematometra was discovered in three out of four teenage girls (75%) reported by Liu, while haematosalpinx was diagnosed in one case. Retrograde menstruation from the rudimentary horn through the ipsilateral fallopian tube could result in the development of a haematosalpinx and endometriosis. Patients with unicornuate uterus are reported to have a signicantly greater prevalence of endometriosis than patients with other uterine anomalies. In the study of Heinonen (7) the frequency of endometriosis was observed to be 21% and all patients had stage I disease, according to ASRM classication (1985). Of the 22 cases of unicornuate uterus with a rudimentary horn reported by Liu (23), endometriosis occurred in seven (31,8%). However, seldom does endometriosis appear in the adolescent age and similarly to haematometra is the result of an obstructive uterine anomaly. Studying the aetiology of endometriosis in adolescence, Liang et al. (24) found endometriosis in three girls with a rudimentary horn, involving the adnexa on the side of the rudimentary horn. In all three cases of the teenage girls with a rudimentary horn and haematometra reported by Liu (23), endometriosis was also evident. Co-existence of haematometra and endometriosis was also observed in the young women with noncommunicating horns reported by Nezhat and Smith and Lin et al. (20,21). When patients with unicornuate uterus and a rudimentary horn conceive, the pregnancy usually occurs in the semi-uterus. In that case the pregnancy could be complicated by abnormal presentations or preterm delivery and may end either in normal vaginal delivery or in a caesarean section, as a result of obstetric

Management of a uterine malformation and review virgin girl. However, bimanual examination is not always useful, as the rudimentary horn is often small and not easy to palpate. The exclusion of some useful diagnostic tools, such as transvaginal ultrasonography, hysterosalpingography and hysteroscopy is also a serious limiting factor for an early diagnosis of a unicornuate uterus. All these methods are capable of providing valuable information about the status of the female internal reproductive organs. Transvaginal ultrasound is helpful in some cases to evaluate the presence of a rudimentary horn, especially if it is performed by a specialist familiar with uterine anomalies. This method is also very helpful for diagnosing an ectopic pregnancy situated in the rudimentary horn. Hysterosalpingography is capable of revealing a semi-uterus with its ipsilateral tube, but it is not able to detect the presence or absence of a rudimentary horn. Hysteroscopy can raise the specialists suspicion when only a single tubular cavity can be seen. In a case of a communicating rudimentary horn, a small contralateral opening of the communicating channel can also be found. Despite those diagnostic limitations, the diagnosis of a uterine anomaly and the precise elucidation of its type, as early as the adolescent age, are of the utmost importance, in order to prevent future problems caused by the anomaly. A careful investigation of the clinical history of the teenager is of great importance, paying special attention to a dysmenorrhoea since menarche with increasing severity. In our case, transabdominal ultrasonography suggested the presence of a uterine anomaly. The diagnosis was conrmed using MRI and laparoscopy, which also elucidated the type of the anomaly and revealed the relevant complications caused by the specic anomaly, such as peritoneal endometriosis, haematometra and haematosalpinx. MRI offers a non-invasive approach towards evaluating the internal and external morphology of the uterus. Pellerito et al. (32) reported a 100% accuracy of the method in assessing women with a surgically proven uterine anomaly; the results were compared to hysteroscopy and laparoscopy. Fedele et al. (33) reported 100% sensitivity and 79% specicity in the diagnosis of congenital uterine anomalies. MRI seems a relatively sensitive diagnostic tool and some authors suggest that it could replace invasive procedures, such as laparoscopy, for the diagnosis of a congenital anomaly of the uterus (34). Furthermore, it provides the possibility to evaluate the renal system. Disadvantages of the method are that is relatively expensive in comparison with other diagnostic tools and is not available in the ofce setting (13). Laparoscopy allows for the evaluation of the external morphology of the uterus. It also offers the added

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advantage of concurrent treatment, as in our case. The main disadvantage of laparoscopy is the invasiveness of the procedure, which is mainly performed under general anaesthesia. According to some authors, the combination of hysteroscopy and laparoscopy is the gold standard in the evaluation of congenital uterine anomalies (3538,12). The exclusion of hysteroscopy in the evaluation of a uterine anomaly in a teenage virgin girl makes the diagnosis difcult. However, it seems that a combination of MRI and laparoscopy in that case provides a precise and sensitive means for diagnosing and also managing a unicornuate uterus with a rudimentary horn. Removal of the rudimentary horn and its ipsilateral tube upon diagnosis as early as the adolescent age is recommended, in order to relieve the symptoms of dysmenorrhoea and lower abdominal pain and to avoid serious future complications, such as endometriosis, infertility, ectopic pregnancy and rudimentary horn rupture. In recent years, laparoscopy has become a viable alternative to laparotomy for the management of uterine congenital anomalies. A review of the literature and our case prove that the excision of a symptomatic noncommunicating rudimentary horn and the ipsilateral fallopian tube is possible by laparoscopic means, even if they have been complicated by haematometra, haematosalpinx and ectopic pregnancy. There are fewer than 35 published cases describing laparoscopic resection of a rudimentary uterine horn. Laparoscopic management of such cases has been reported by Canis et al., Nezhat et al., Mais et al., Kadir et al., Falcone et al., Giatras et al., Perrotin et al., Durin et al., Theodoridis et al. (3948). Cases of laparoscopic resection of a pregnant rudimentary horn have been published by Dicker et al., Park and Dominguez, and Henriet et al. (24,49,50). Finally, laparoscopic management of a rudimentary noncommunicating uterine horn and its complications has been performed hitherto in only a few teenage girls, with a promising outcome. Cases of operative laparoscopy in such anomalies during adolescence have been reported by Amara et al., Nezhat and Smith, Saleh et al., and Spitzer et al. (5153). In conclusion, a unicornuate uterus with a noncommunicating rudimentary horn is a rare congenital anomaly of the female genital tract and is hard to diagnose before clinical manifestation, especially in young women. Most often the diagnostic procedure begins after the occurrence of complications, such as haematometra, haematosalpinx and endometriosis, which cause acute symptoms such as dysmenorrhoea and lower abdominal pain. The combination of MRI and laparoscopy seems to be an effective and precise tool in the diagnosis of a unicornuate uterus with a

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rudimentary horn in teenage girls. In order to prevent further and more severe complications, such as infertility, ectopic or rudimentary horn pregnancy and rudimentary horn rupture, an early excision of the horn and its ipsilateral tube, even during the adolescent age, is indicated. According to our opinion, operative laparoscopy is an excellent alternative treatment for the symptomatic noncommunicating rudimentary horn. Commonly accepted benets of the method, such as improved visualization of the Douglas pouch, smaller incisions, decreased adhesion formation, reduced postoperative pain and shorter hospital stay, are in favour of a laparoscopic approach. Other factors that should be considered are availability of proper instrumentation and surgical experience. Declaration of interest: The authors report no conicts of interest. The authors alone are responsible for the content and writing of the paper.

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