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Congenital Anomalies 2007; 47, 156–157
doi:10.1111/j.1741-4520.2007.00163.x
CASE REPORT
Unruptured term pregnancy with a live fetus with placenta percreta in a non-communicating rudimentary horn Sharda Patra, Manju Puri, Shuba Sagar Trivedi, Reena Yadav, and Jyoti Bali Department of Obstetrics and Gynecology, Lady Hardinge Medical College & Smt. S.K. Hospital, New Delhi, India
ABSTRACT Pregnancy in a non-communicating rudimentary horn is rare and such a pregnancy culminating in the delivery of a live fetus is even rarer. Despite advances in ultrasonography, the accuracy of ultrasound in diagnosing rudimentary horn pregnancy at advanced gestation remains elusive. Confirmatory diagnosis is made only at laparotomy. We report a multigravidae who presented at 37 weeks with transverse lie oligoamnios and decreased perception of fetal movement since quickening. Laparotomy for placenta accreta suspected on ultrasound revealed non-communicating unruptured rudimentary horn pregnancy with a live fetus and placenta percreta. Successful extraction of a term live fetus weighing 2.7 kg with excision of the rudimentary horn was carried out. Key Words: non-communicating, placenta accreta, pregnancy, rudimentary horn
INTRODUCTION Pregnancy in a rudimentary uterine horn is a rare condition. Such pregnancies rarely reach viability and often result in rupture of the horn, causing significant fetal and maternal morbidity and mortality (O’Leary & O’Leary 1963). In 80% of cases, the uterine rupture occurs in the first and second trimester, and in the remaining 20% in the third trimester (Nahum 2002). Rudimentary horn pregnancies reaching term with birth of a live fetus are very rare (Jayasinghe et al. 2005; Pal et al. 2006; Poonam et al. 2007). We report an interesting case where pregnancy in a noncommunicating rudimentary horn with placenta percreta reached 37 weeks without rupture and resulted in the birth of a healthy live baby.
CASE REPORT A 25-year-old gravida 3, para 2 with previous normal vaginal deliveries was admitted from the antenatal clinic at 37 weeks’ gestation with transverse lie and gross oligohydramnios. She was an unbooked case with an uneventful pregnancy except that she perceived less fetal movements in this pregnancy compared to her previous two pregnancies. On examination her vitals were normal. Obstetrical examination revealed a markedly dextrorotated uterus with fundal height corresponding to 32 weeks’ pregnancy. Liquor was reduced and fetal parts were identified with difficulty. The lower pole of the uterus was empty. Ultrasound examination Correspondence: Sharda Patra M.D., Department of Obstetrics and Gynecology, Lady Hardinge Medical College & Smt. S.K. Hospital, New Delhi 110001, India. Email:
[email protected] Received May 23, 2007; revised and accepted August 9, 2007.
© 2007 The Authors Journal compilation © 2007 Japanese Teratology Society
showed a single live fetus in transverse lie with fetal parameters corresponding to 36–37 weeks’ gestation. Liquor was markedly reduced (AFI 3) and the placenta was covering the os. There was loss of integrity of interface between placenta and myometrial wall. A provisional diagnosis of central placenta previa with accreta was made and subsequently confirmed on Doppler flow studies. The patient was posted for elective ceserian section. When the abdominal cavity was opened a term baby was found lying in the rudimentary horn, which was attached to a bulky unicornuate uterus on the left side by a band of fibrous tissue 2–3 cm thick. The tube attached to the rudimentary horn was stretched out but normal looking. The ovary was seen posterior to the horn. The wall of the rudimentary horn was thinned out with large vessels running over its surface and oozing blood from the surface. Omentum was adherent to its anterior surface. The placental tissue was seen through the thinned anterior wall of the horn. A live male baby of 2.7 kg, with an Apgar score of 9 was extracted through an incision on the anterior wall of the horn cutting through the placenta. The placenta could not be separated from the wall of the horn. There was no communication of the horn with the uterus. The rudimentary horn and the placenta was excised. Right-sided tubal ligation was performed. Histopathology of the specimen confirmed the diagnosis of placenta percreta (Fig. 1). The postoperative period was uneventful.
DISCUSSION The estimated incidence of rudimentary uterine horn pregnancy is from one in 76 000 to one in 140 000 (McCarthy 1999; Nahum 2002). Rudimentary horn is the result of an incomplete fusion of the mullerian elements. In 83–90% of cases, there is no direct communication between the horn and the functional portion of the uterus and fertilization is thought to occur via transperitoneal migration of the sperm (O’Leary & O’Leary 1963). The most serious complication of rudimentary horn pregnancy is rupture of the horn which occurs because of the ill-developed endometrial lining and musculature that fails to accommodate a full-term pregnancy (Nahum 2002; Jayasinghe et al. 2005) The first case of uterine rupture associated with a rudimentary uterine horn was reported in 1699 (Mauriceau F, 1721). The maternal mortality rate before the advent of ultrasonography was as high as 47.6%. Rupture of the horn is still common but no case of maternal death has been published since 1960 (Nahum 2002). Rupture can occur into the abdominal cavity or into the unicornuate uterus. The average duration of gestation has been estimated at 21.5 weeks (Nahum 1997). Since 1946 live births have been reported in only 12% of cases published. Diagnosis of rudimentary horn pregnancy should be made early in pregnancy (Rolen et al. 1966) Sonographic diagnosis of a rudimentary uterine horn during pregnancy remains elusive because the enlarging horn with a thinning myometrium can often obscure the anatomic structures and
Unruptured term rudimentary horn pregnancy
Placenta percreta Streched left tube and ovary Round ligament Fig. 1
Laparotomy findings. Unicornuate uterus with rudimentary horn pregnancy.
mimics conditions like bicornuate uterus, interstitial pregnancy and abdominal pregnancy. The sensitivity of ultrasound examination for diagnosis has been reported as 26% and the sensitivity decreases as the pregnancy advances (Jayasinghe et al. 2005). In our patient, rudimentary horn pregnancy was missed on ultrasonography. Published reports show very low preclinical (8%) and preoperative detection rates (29%) (Jayasinghe et al. 2005). This highlights the importance of a careful ultrasound examination in the first and early second trimester, where emphasis should be not only on the details of pregnancy but also on detection of uterine anomaly (Daskalakis et al. 2002). Tsafrir et al. (2005) suggested the following criteria for sonographic diagnosis of rudimentary horn pregnancy: (i) a pseudo pattern of an asymmetrical bicornuate uterus; (ii) absent visual continuity of tissue surrounding the gestational sac and the uterine cervix; and (iii) the presence of myometrial tissue surrounding the gestational sac. Additionally, MRI can be used to confirm the diagnosis before an invasive procedure is undertaken (Ozeren et al. 2004). Live births are very rare in this condition and until 1999 only 13 fetal survivals were reported (McCarthy 1999). In a review from 1990 to 1999 only 6% of rudimentary horn pregnancies were reported to have progressed to term with 13% neonatal survival and this was because of early detection and intervention (Shin & Kim 2005; Shinohara et al. 2005). The most noticeable item in our
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patient was the decreased perception of fetal movements throughout the pregnancy. Therefore, any pregnancy with similar complaints coupled with features such as abnormal uterine contour, fetal malpresentation and oligohydramnios should be viewed with suspicion of an abnormal pregnancy.
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© 2007 The Authors Journal compilation © 2007 Japanese Teratology Society
