Mohammad Faizul Suhail, MD, Hina Khan,MD, and Safia Suhail,MD, are from the
City Nursing and Maternity Home, Allahabad-211003, India.
Correspondence: Dr. Safia Suhail, Senior Gynecologist and Director, City Nursing and Maternity Home, 21, Minhajpur, Leader Road, Allahabad-211003, India.
Abstract: When paramesonephric ducts fuse incompletely at the level of the uterine fundus, a bicornuate uterus is formed; a muscular uterine septum is also present, and classification depends on the extent of this septum. A variant of the complete form of bicornuate uterus is the bicornuate bicollis uterus, in which the septum extends to the external os. In the present case of bicornis bicollis uterus, the pregnant woman delivered a male viable baby with breech presentation by LSCS. To our knowledge, such uterine anomaly has only been sporadically reported as revealed by our survey on bicornis bicollis uterus in the literature for the last over nine decades. We here report a case.
Key Words: uterine anomaly, bicornis-bicollis, breech presentation
Incomplete fusion of the Mullerian or paramesonephric ducts results in the most common types of uterine malformation: uterus didelphys, uterus bicornis bicollis, uterus bicornis unicollis, uterus subseptae, uterus arcuatus, uterus unicornis. Uterus bicornis bicollis1 is characterized by double or single vagina, double cervix and two single-horned uteruses which show partial fusing of their muscular walls with duplication running right down to the uterine orifice.
Many complications can occur: Fertility is usually unaffected but complications may arise during labor: late abortion due to poor implantation and/or premature labor, increased sensitivity to stimuli, malpresentation – especially in uterus subseptus, obstructed labor – if one horn of a bicornute uterus, especially a bicornute, bicollis uterus, falls behind the horn in which the conception sac has grown, retained placenta, postpartum hemorrhage.
Pregnancy with a bicornuate uterus has to be considered at risk since there is a higher risk of complications and because the infant may suffer congenital defects, premature birth and with intrauterine growth retardation2. Women with congenital uterine malformation usually have higher incidence of infertility and complications during pregnancy and delivery. Bicornuate uterus could be associated with poorer obstetric outcome3. Offspring of mothers with bicornuate uterus had a four times higher risk of congenital defects than infants born to women with normal uteri. The risk was statistically significant for some specific defects such as nasal hypoplasia, omphalocele, limb deficiencies, teratomas, and acardiaanencephaly4.
Twenty six year old Rabab Fatima, a primipara having fullterm pregnancy with labor pain and leaking per vaginum, was admitted on 2nd Oct. 2007 with BP 126/68 mm Hg, pulse 70/min, Hb10.8 gm%, and blood group B+ve. Urine sample showed absence of albumin and sugar.
Fig 1: Bicornuate bicollis uterus, with gravid right horn; groove between the 2 horns appreciated
Fig. 2: Biconuate bicollis uterus with right and left fallopian tubes and ovaries
Fig. 3: Two separate uterine cavities visualised with finger in left cavity
Fig. 4: Thick muscular septum (held by artery forceps) separating two cavities and extending to the cervix.
Per abdominal examination revealed a fetal height of 36 weeks, breech presentation, and presence of fetal heard sounds. PV examination showed two vaginal openings separated by a thick wall (vaginal duplex), two cervixes (bicervical; cervix two fingers loose, taken up, breech presentation, and membrane absent.
LSCS was for breech presentation. Abdomen was opened by transverse section, bladder pushed down, uterus opened by transverse section and a male baby, weighing 2.6 kg, was delivered by breech on the same day. The baby cried immediately after delivery and was in good condition. Placenta was delivered and uterine cavity cleaned.
Uterus, on examination, was found to have another horn on the left side with a tube and ovary on its left side, besides the one that was gravid (right horn, with a tube and ovary attached to it on right side).
The well-contracted uterus was closed in layers; PPH was ruled out. Abdominal cavity was cleaned and closed in layers. Wound was cleaned and dressed.
Post-operative period was uneventful. Recovery was fair. Stitches were removed; prognosis was good. The patient was discharged on the 8th day. This women having two horns, two cervixes, two vaginal openings separated by a thick muscular wall Thus this woman is having uterine anomaly of bicornis bicollis uterus. She had pregnancy in her right horn.
The occurrence of a pregnancy in bicornis bicollis uterus has been reported only sporadically in the literature. Our first case in 27 years is reported here. The detection of congenital uterine anomalies will increase because of heightened physician awareness and improved diagnostic modalities.
The authors have gone through literature on bicarnuate uterus specially bicollis for the last over nine decades which shows sparingly reported condition. Ludmir et al.5 conducted studies for 8 years managing 42 women with 101 pregnancies with previously diagnosed but uncorrected uterine malformations referred to their institution for high-risk obstetric care. The population studied consisted of four groups of pregnancies with uterine anomalies as unicornuate (5), bicornuate (61), septate (25), and didelphys (10). Sixty percent of the pregnancies in the unicornuate and didelphys group reached term, 39% in the bicornuate group, and 48% in the septate group.
They suggest that high-risk obstetric intervention does not obviate the potential benefit of metroplasty, especially for patients with a bicornuate uterus and believe that traditional indications for metroplasty should continue to be used.
Acién6 studied 176 patients with uterine malformations including bicornuate (n = 49) and bicornis-bicollis (n = 17) and reported that patients with uterine malformations have higher rates of reproductive loss, pre-term delivery, breech presentation and complications that increase obstetric intervention and perinatal mortality. Zanetti et al.7 analyzed a series of 13470 hysterosalpingographic investigations which demonstrated congenital uterine malformations in 1160 cases (8.6%) and uterus unicornis unicollis and uterus bicornis unicollis together accounted for over a quarter.
Ben-Rafael et al.8 evaluated the incidence of pregnancy-induced hypertension (PIH) in women with congenital uterine malformations by examining the pregnancy complications of 67 women with uterine anomalies compared with a control group of 130 women with normal-shaped uterus and reported a significantly increased (p less than 0.04) rate of PIH in women with uterine malformation as well as a 2-fold higher frequency of preeclampsia.
In 1914, Marshall9 reported a case of Uterus Bicornis Bicollis with six months’ pregnancy right horn and, pyometra left horn. In this case pregnancy of only six months was reported along with pyometra in left horn where as in the present case a full term delivery by cesarian section was performed and the left horn was normal. Interestingly, pregnancy has been found in right horn. In case of single pregnancy, it is in the right uterus in uterus didelphys, and even in the present case of bicornuate uterus, pregnancy has developed preferentially in the right horn. Why pregnancy happens preferably in the right uterus or right horn has been discussed elaborately in our previous communication on uterus didelphys case report10.
Congenital uterovaginal anomalies can have adverse effects on pregnancy outcome. Early diagnosis and an aggressive evaluation of any patient presenting with mid-trimester abortion, premature labor, malpresentation, or retained placenta may prevent additional pregnancy wastage and maternal morbidity. With timely and accurate diagnosis, appropriate management is likely to provide the best possible outcome for all such patients.