Published on March 10, 2014
Ruptured ectopic pregnancy in non-communicating right rudimentary horn: A case report
Case Report Ruptured ectopic pregnancy in non-communicating right rudimentary horn: A case report Ahmed S. Elagwany*, Hisham H. Elgamal, Tamer M. Abdeldayem Department of Obstetrics and Gynecology, Alexandria University, Egypt a r t i c l e i n f o Article history: Received 19 August 2013 Accepted 4 October 2013 Available online xxx Keywords: Acute abdomen Hemoperitoneum Rudimentary horn Rupture Ultrasound a b s t r a c t Rudimentary horn is a developmental anomaly of the uterus, and pregnancy in a non- communicating rudimentary horn is very difﬁcult to diagnose before it ruptures. As the fetus enlarges in the rudimentary horn, the chances of rupture in the ﬁrst or second trimester are increased. Catastrophic hemorrhage results in increased maternal and perinatal mortality and morbidity. To date, management of such cases remains a challenge due to diagnostic dilemma. Expertise in ultrasonography and early resort to surgical management are lifesaving in such cases. A case of undiagnosed rudimentary horn preg- nancy presented to our department in shock with features of acute abdomen, and the diagnosis was conﬁrmed at laparotomy that revealed ruptured rudimentary horn preg- nancy. And excision of the accessory horn was done. Copyright ª 2013, Indraprastha Medical Corporation Ltd. All rights reserved. 1. Introduction Congenital uterine malformations are the result of abnormal Mullerian duct development, fusion, canalization and septal resorption. The prevalence of congenital uterine anomalies among fertile women is reported as 1:200e1:600. Where there is atresia of one of the Mullerian duct, there is a unicornuate uterus with a single tube. Its true incidence is unknown and is approximately estimated at a rate of about 1 in 250. Unicornuate uterus may also have a rudimentary horn. Rudimentary horn is one of the rarest congenital uterine anomalies. The prevalence of unicornuate uterus with rudimentary horn is even rarer i.e. 1:100,000.1 The rudimentary horn may consist of a functional cavity, or it may be a small solid lump of the uterine muscle with no functional endometrium. In a series of 366 cases with rudi- mentary horn, non-communicating horn accounted for 92% of cases, and renal anomaly was found in 36% cases. Unilateral renal agenesis was found in 38% cases in another series.2 Provided there is no obstruction to menstrual ﬂow, these uterine anomalies present few problems in the absence of pregnancy. Pregnancy in a rudimentary horn is rare. The incidence of rudimentary horn pregnancy is difﬁcult to calculate. Frequently quoted ﬁgures are between 1 per 76,000 and 1 per 140,000 pregnancies.3 A case of pregnancy occurring in a rudimentary horn with consequent rupture is reported. 2. Case report An unusual case of a 25-year-old woman, G1P0, with a preg- nancy of 18 weeks, presented to the emergency unit of our * Corresponding author. El-Shatby Maternity Hospital, Alexandria University, Alexandria, Egypt. Tel.: þ20 1228254247. E-mail address: Ahmedsamyagwany@gmail.com (A.S. Elagwany). Available online at www.sciencedirect.com journal homepage: www.elsevier.com/locate/apme a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1 e4 Please cite this article in press as: Elagwany AS, et al., Ruptured ectopic pregnancy in non-communicating right rudimentary horn: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.10.002 0976-0016/$ e see front matter Copyright ª 2013, Indraprastha Medical Corporation Ltd. All rights reserved. http://dx.doi.org/10.1016/j.apme.2013.10.002
department with acute abdominal pain and hypovolemic shock. The patient did not seek antenatal care before admis- sion. Pregnancy was diagnosed by urine pregnancy test after a missed period for one week. Ultrasound was not done till admission .The patient was illiterate and living in a rural area far away from any health facility. She was not complaining so she did not seek medical advice. No other signiﬁcant his- tory was noted. On examination, the patient was drowsy, in agony and in a nearly shock state with pale, cold, clammy extremities, a thready pulse of 120 beats/minute and arterial blood pressure of 70/40 mmHg. Abdominal examination revealed distention and tenderness all over. On vaginal examination, mild vaginal bleeding was noted. A speculum examination did not reveal any cervical or vaginalpathology.The cervicalos was tightlyclosed. The patient was resuscitated with intravenous ﬂuids and blood transfusion. Further assessment with transabdominal ultrasound showed a free ﬂoating nonviable fetus of 18 weeks gestation in abdominal cavity with anhydramnios and moderate abdom- inal collection surrounding. Absent visual continuity of myo- metrial tissue surrounding the fetus was noted. The placenta was intrauterine, lying posterior with no retroplacental clots seen. Abdominal tapping with spinal needle revealed non clotted dark bloody collection. In view of the previous data, a provisional diagnosis of a ruptured uterus with a differential diagnosis of an abdominal pregnancy was made. An emergency laparotomy was performed immediately after resuscitation. Intraoperative ﬁndings revealed a uni- cornuate uterus with a ruptured pregnancy in a right rudi- mentary horn. Also, a deadfetus ﬂoating inthe peritonealcavity with blood clots surrounding was noted. The placenta was still attached to the cavity of the rudimentary horn (Figs. 1 and 2). It was, however, difﬁcult to visualize the uterine septum. No communication from the horn to the contralateral uterine cavity was seen. The uterus, lying separate in the pelvis, was soft in consistency, globular and enlarged to a size consistent with ten weeks. The right fallopian tube and ovary were found healthy and were attached normally to the unicornuate uterus. Excision of the rudimentary horn and the right fallopian tube with conservation of the right ovary was done together with the nonviable fetus and the placenta. All the pedicles were secured with good hemostasis. A total volume of nearly 2 L of hemoperitoneum blood was also removed. Both the ureters and kidneys were normal. Due to the signiﬁcant blood loss throughout the operation, the patient was given a massive blood transfusion of four units and two units of plasma transfusion. The excised specimen was sent for histopathological ex- amination, which was reported as (sections from the uterine horn show areas of hemorrhage and necrosis. Section from the fallopian tube was morphologically normal. Sections of the placenta show ﬁbrosed chorionic villi with syncytial knots. No villitis was seen). Histopathology conﬁrmed the diagnoses (ruptured pregnant uterine rudimentary horn). Post-operatively, hemoglobin level was 9.8 g/dl. Patient had a smooth postoperative recovery. The patient was counseled for contraception with oral pills for one year. The patient was discharged healthy two days after admission. Follow-up appointment was planned for intravenous uro- gram to rule out any associated renal anomalies. No associ- ated renal anomaly was diagnosed. The patient was Fig. 1 e A nonviable fetus with placenta attached to ruptured right rudimentary horn. a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1 e42 Please cite this article in press as: Elagwany AS, et al., Ruptured ectopic pregnancy in non-communicating right rudimentary horn: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.10.002
scheduled for a hysterosalpingogram 6 weeks post- operatively before any further decisions on future pregnancy. 3. Discussion The incidence of uterine anomalies ranges between 0.5% and 2%. Unicornuate uterus (Type II) can further be classiﬁed as those having an endometrial cavity and those with no endo- metrial cavity, and 90% of the cases are those with an endo- metrial cavity. A further division can be between the communicating and non-communicating types. Identiﬁcation of unicornuate uterus is almost always accidental, as it is usually asymptomatic until reproductive age. Initial presen- tation often takes place with a recurrent ﬁrst trimester miscarriage (5e10%), a second trimester loss (25%), or during an infertility work-out.4 Mariceau and Vassal published the ﬁrst description of a rudimentary horn pregnancy in 1669, and 600 cases have since been described.5 Pregnancies occur in both communicating and non-communicating horns in proportion to their relative incidence and are equally likely to rupture. Neonatal mortality is very high as most cases are emergency laparotomies after uterine rupture at premature gestational age. Maternal mor- tality is low (0.5%) but morbidity is very high in view of massive blood loss and morbidly adherent placentation.2,6 In cases with successful pregnancy, the risks of recurrent early trimester loss and mid-trimester loss are increased, with preterm delivery and abnormal fetal lie contributing factors. Pregnancy in a rudimentary horn is a form of ectopic pregnancy and it can cause mortality or severe morbidity. It can occur in both communicating and non-communicating types. In the case of a non-communicating rudimentary horn like our patient had, it is postulated that the fertilization was possibly due to transperitoneal migration of the sperm.7 The outcomes of pregnancy in rudimentary horns are ectopic pregnancy (2.7%), ﬁrst trimester loss 24.3%, second trimester abortion 9.7%, preterm delivery 20.1%, intrauterine demise (10.5%) and 49.9% live birth up to 28e30 weeks of gestation. Uterine rupture in the second trimester is usually a life-threatening condition resulting from a much thinner myo- metrial part of the rudimentary horn, with the non-functional endometrium leading to placenta adherence anomalies.8 The prerupture diagnosis of pregnancy in rudimentary horn has drastically reduced maternal mortality.7 But the sensitivity of ultrasound to detect prerupture rudimentary horn pregnancy is very poor (30%),8 probably because of rarity of the diagnosis and non-familiarity of the radiologists about this potentially lethal condition. Early diagnosis before rupture can be managed laparoscopically.9 Prerupture diagnosis of rudimentary horn pregnancy is challenging. A careful ultrasound in the ﬁrst trimester with a high index suspicion, one should be able to make a diagnosis of pregnancy in the rudimentary horn and sensitivity de- creases as the pregnancy advances.10 In such cases magnetic resonance imaging (MRI) is very useful not only in conﬁrming the diagnosis, it also helps to plan the surgery.11 Tubal preg- nancy, cornual pregnancy, and abdominal pregnancy are common sonographic and clinical misdiagnosis. It is very difﬁcult to establish diagnosis in second trimester due to lack of deﬁnitive clinical criteria. Tsafrir et al. proposed the following criteria for ultrasono- graphic diagnosis: 1. a pseudo pattern of an asymmetrical bicornuate uterus, 2. absent visual continuity tissue sur- rounding the gestation sac and the uterine cervix, and 3. the presence of myometrial tissue surrounding the gestation sac.8 Three-dimensional ultrasound imaging and MRI are useful tools in the improvement of diagnostic accuracy, guiding both counseling and surgical planning.12 The traditional and established treatment for rudimentary horn pregnancy is surgical removal of the pregnant horn even in unruptured case to prevent rupture and recurrent rudi- mentary horn pregnancy.13,14 Laparoscopic excision of the rudimentary horn pregnancy prior to rupture has been done successfully since last two de- cades.10,11 Renal anomalies are found in 36% of cases; hence it is mandatory to assess these women prior to surgery and if required better to do ureterolysis before the excision of the horn. Medical management with methotrexate during early preg- nancy in the rudimentary horn has also been used successfully.14 In our patient, the diagnosis of ectopic pregnancy was only established when she went into hypovolemic shock at 18 weeks. The non-communicating rudimentary horn rupture was only conﬁrmed intraoperatively and removal of the rudimentary horn and ipsilateral fallopian tube was done. This was to reduce the risk of the patient having another ectopic pregnancy in the future. This case further raises the question of whether routine excision of rudimentary horn be undertaken in women with Fig. 2 e Site of perforation in right rudimentary horn. a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1 e4 3 Please cite this article in press as: Elagwany AS, et al., Ruptured ectopic pregnancy in non-communicating right rudimentary horn: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.10.002
unicornuate uterus as a prophylaxis to prevent such catas- trophes. In our opinion, routine laparoscopic excision of rudimentary horn with ipsilateral fallopian tube should be offered to these women and those refusing should be adequately counseled regarding the potential complications and if pregnancy occurs in rudimentary horn ﬁrst trimester laparoscopic excision should be done. 4. Conclusion Rudimentary horn pregnancy is a rare complication which carries grave risk to the mother. More than 90% of the cases present in second trimester with intraperitoneal hemorrhage due to rupture of the horn. Ideally, history of repeated abor- tions must be investigated before conception to exclude Mullerian duct malformation. In addition, an intravenous pyelogram is indicated because of high incidence of associ- ated urinary anomalies in the presence of genital anomalies. Diagnosis prior to rupture should be the concern in early pregnancy with either ultrasound or MRI to prevent life- threatening complications. The recent trend is to do laparo- scopic excision of the rudimentary horn; laparotomy is still an option when the patient is in shock. Conﬂicts of interest All authors have none to declare. r e f e r e n c e s 1. Grimbizis GF, Camus M, Tarlatzis BC. Clinical implications of uterine malformations and hysteroscopic treatment results. Hum Reprod Update. 2001;7:161e174. 2. Jayasinghe Y, Rane A, Stalewski H. The presentation and early diagnosis of the rudimentary uterine horn. Obstet Gynecol. 2005;105:1456e1467. 3. Acien P. Incidence of Mullerian defects in fertile and infertile women. Hum Reprod. 1997;12:1372e1376. 4. Fitzmaurice LE, Ehsanipoor RM, Porto M. Rudimentary horn pregnancy with herniation into the main uterine cavity. Am J Obstet Gynecol. 2010;202(3). 5. Chopra S, Keepanasseril A, Rohilla M. Obstetric morbidity and the diagnostic dilemma in pregnancy in rudimentary horn: retrospective analysis. Arch Gynecol Obstet. 2009;280:907e910. 6. Lawhon BP, Wax JR, Dufort RT. Rudimentary uterine horn pregnancy diagnosed with magnetic resonance imaging. Obstet Gynecol. 1998;91(5 Pt 2):869. 7. Nahum GG. Rudimentary uterine horn pregnancy. The 20th- century worldwide experience of 588 cases. J Reprod Med. 2002;47:151e163. 8. Tsafrir N, Rojansky HY, Sela J. Rudimentary horn pregnancy: ﬁrst-trimester prerupture sonographic diagnosis and conﬁrmation by magnetic resonance imaging. J Ultrasound Med. 2005;24:219e223. 9. Yan CM. Laparoscopic management of three rare types of ectopic pregnancy. Hong Kong Med J. 2010;16:132e136. 10. Sharma D, Usha MG, Gaikwad R. Laparoscopic resection of unruptured rudimentary horn pregnancy. Int J Reprod Contracept Obstet Gynecol. 2013;2:95e98. 11. Edelman AB, Jensen JT, Lee DM. Successful medical abortion of a pregnancy within a noncommunicating rudimentary uterine horn. Am J Obstet Gynecol. 2003;189:886e887. 12. Nahum G, Stanislaw H, McMohan C. Preventing ectopic pregnancies: how often does transperitoneal transmigration of sperm occur in effecting human pregnancy? Br J Obstet Gynecol. 2004;111:706e714. 13. Reichman D, Laufer MR, Robinson BK. Pregnancy outcomes in unicornuate uteri: a review. Fertil Steril. 2009;91:1886e1894. 14. Sunilkumar KS, Yaliwal LV, Amarnath A. Ruptured rudimentary horn of the unicornuate uterus at 16 weeks of pregnancy: a case report. Int J Reprod Contracept Obstet Gynecol. 2013;2:248e250. a p o l l o m e d i c i n e x x x ( 2 0 1 3 ) 1 e44 Please cite this article in press as: Elagwany AS, et al., Ruptured ectopic pregnancy in non-communicating right rudimentary horn: A case report, Apollo Medicine (2013), http://dx.doi.org/10.1016/j.apme.2013.10.002
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