Diagnosis and management of pre-rupture stage of the pregnant horn are difficult and usually missed on a routine ultrasound scan. Also most cases are detected after rupture of pregnant horn. We presented a 28-year-oldG2 L1 woman with diagnosis of rudimentary horn pregnancy (RHP) at 14 weeks of gestation. We diagnosed her with a normal intrauterine pregnancy, whereas a pregnancy in a right-sided non-communicating rudimentary horn with massive he-moperitoneum was later discovered on laparotomy. RHP has a high risk of death for mother, so there must be a strong clinical suspicion for the diagnosis of RHP. Although there is a major advancement in field of diagnostic ultrasound and other imaging modalities, prenatal diagnosis has remained elusive and a laparotomy surgery is considered as a definitive diagnosis. Â© 2017, Royan Institute (ACECR). All rights reserved

Forghani, F.

Kaveh, M.

Mehdizadeh Kashi, A.

Sadegi, K.

eprints Iran University of Medical Sciences

Case Report318Pregnancy in Non-Communicating Rudimentary Horn of A Unicornuate UterusMania Kaveh, M.D.1, 2, Abolfazl Mehdizadeh Kashi, M.D.1, Kambiz Sadegi, M.D.3, 4, Forough Forghani, M.D.2* 1. Endometriosis and Gynecological Disorder Research Center, Iran University of Medical Science, Tehran, Iran2. Department of Obstetrics and Gynecology, Zabol University  of Medical Science, Zabol, Iran3. Pain Research Center, Iran University of Medical Science, Tehran, Iran4. Department of Anesthesiology, Zabol University  of Medical Science, Zabol, IranAbstract Diagnosis and management of pre-rupture stage of the pregnant horn are difficult and usually missed on a routine ul-trasound scan. Also most cases are detected after rupture of pregnant horn. We presented a 28-year-oldG2 L1 woman with diagnosis of rudimentary horn pregnancy (RHP) at 14 weeks of gestation.  We diagnosed her with a normal intrauterine pregnancy, whereas a pregnancy in a right-sided non-communicating rudimentary horn with massive he-moperitoneum was later discovered on laparotomy. RHP has a high risk of death for mother, so there must be a strong clinical suspicion for the diagnosis of RHP. Although there is a major advancement in field of diagnostic ultrasound and other imaging modalities, prenatal diagnosis has remained elusive and a laparotomy surgery is considered as a definitive diagnosis.Keywords: Pregnancy, Rudimentary, UterusCitation: Kaveh M, Mehdizadeh Kashi A, Sadegi K, Forghani F. Pregnancy in non-communicating rudimentary horn of a unicornuate uterus. Int J Fertil Steril. 2018; 11(4): 318-320. doi: 10.22074/ijfs.2018.5022.Introduction Rudimentary horn pregnancy (RHP) as a rare incidence, has been estimated at 1:76,000-1:160,000 pregnancies (1).  It has also been reported that 75-83% of cases are the pregnancy in non-communicating rudimentary horn that is  caused by transmigration of peritoneal sperm or fertilized ovum (2). Gynecological and obstetrical com-plications of pregnancy in unicornuate uterus with a ru-dimentary horn are as following: i. Spontaneous abortion, ii. Preterm labor, iii. Infertility, iv. Endometriosis, v. He-matometra, vi. Intrauterine growth restriction (IUGR), vii. Intra peritoneal bleeding, and viii.. Uterine rupture. Kidney abnormalities have also been reported in 31% of cases, while the patients were diagnosed after reaching their stable condition (3). Rupture of RHP is considered as a life threatening condition for mothers. We used time-ly laparotomy, excision of the horn and blood transfusion to save a 28-year-old G2 L1 woman who was initially di-agnosed with a normal intrauterine pregnancy, but a 14-week pregnancy in a right-sided non-communicating ru-dimentary horn with massive hemoperitoneum was later discovered on laparotomy.Case reportA 28-year-old G2L1 woman who was 14 weeks preg-nant was admitted at Amiralmomenin Hospital, Zabol, Iran, in November 2015, with generalized abdominal pain, nausea and vomiting.  The patient who had a pre-Received: 3 Aug 2016, Accepted: 14 Mar 2017*Corresponding Address: P.O.Box: 14977456367, Department of Obstetrics and Gynecology, Amiralmomenin Hospital, Zabol University of Medical Sci-ence, Zabol, IranEmail: forough-iran2005@yahoo.comRoyan InstituteInternational Journal of Fertility and Sterility Vol 11, No 4, Jan-Mar 2018, Pages: 318-320vious cesarean section received early prenatal care two years ago and an ultrasound exam at 14 weeks of ges-tation (a day before she was admitted at the hospital). The patient suffering from hypovolemic shock, was ex-tremely pale, and had a weak pulse of 120-130 beats per minute and a blood pressure of 80/60 mm Hg. Physi-cal exam revealed impaired consciousness and agita-tion, generalized abdominal tenderness with sharp right lower quadrant (RLQ) pain, no vaginal bleeding, and a closed cervix. A portable ultrasound detected more than 2 liters of free fluid in the abdomen and pelvis that con-firmed the presence of unicorn ate uterus and a 14-week pregnancy in right-sided rudimentary horn. After fluid resuscitation, the patient was transferred to the operating room for an emergency laparotomy. Her blood pressure was 90/60 mm Hg at the time of laparotomy. During laparotomy, we founded that right-sided non-communicating rudimentary horn was already ruptured and the fetus with amniotic sac extruded into the perito-neal cavity with presence of about a 3-liter hemoperito-neum (Figs.1-3). The rudimentary horn was then excised (Fig.4) and the abdomen closed following hemostasis. Fur-thermore, patient received 3 unites of pack cell and recov-ered well after surgery. She was discharged with satisfac-tory condition on fifth post-operative day after the kidney anomalies ruled out. This study was approved by the Ethics Committee of Iran University of Medical Sciences, Tehran, Iran. Written informed consent was obtained from case. Int J Fertil Steril, Vol 11, No 4, Jan-Mar 2018              319Fig.1: Rudimentary horn pregnancy still attached to the main horn. Fig.2: A 4-cm ruptured rudimentary horn with placenta partially protrud-ing from it.'Fig.3: Fetus with placenta.Fig.4: Excised rudimentary horn.DiscussionPregnancy in a non-communicating rudimentary horn is the results of developmental defect of one Müllerian duct or incomplete connection with Müllerian ducts on the opposite site that has been estimated at 1:76,000-1:160,000 pregnancies (1-4). The first case of uterine rupture following RHP was reported by Kanagal and Hanumanalu (5). It has been reported that the timing of a ruptured rudimentary horn that is mainly associated with horn musculature and its ability to hypertrophy is esti-mated between 5 and 35 weeks. The early diagnosis of RHP is likely to prevent maternal morbidity and mortal-ity. The best management strategies for early diagnosis of RHP are as: i. Ultrasound, ii. Hysterosalpingography, hysteroscopy, laparoscopy, as well as iii. Magnetic reso-nance imaging (MRI) (5). It is noted that the sensitivity of ultrasound is 26%, although its sensitivity decreases when the maternal age increases (6). However, RHP is likely to be missed by the most ex-perienced radiologist. The most common ultrasound re-ports that leads to misdiagnose RHP are as follows: i. Tubal pregnancy, ii. Cornal pregnancy, iii. Intrauterine pregnancy, and iv. Abdominal pregnancy (7). It is dif-ficult to confirm a rudimentary horn with thin myome-trium diagnosis because of obscuring adjacent anatomi-cal structures. The following diagnostic criteria for RHP were indicated by Tsafrir et al. (8) using ultrasonogra-phy: i. Pseudo pattern of asymmetrical bicornuate uter-us, ii. Non-continuity between tissue surrounding the gestational sac and the uterine cervical canal, and iii. The presence of myometrial tissue surrounding the ges-tational sac.  Furthermore, a hyper vascularization pat-tern like placenta accrete is considered as an indication for the diagnosis of RHP that is detected by both Color Doppler ultrasound and Doppler ultrasound. Samuels and Awonuga (9) have reported a uterine rupture after labor induction with misoprostol.The application of different methods of labor induc-tion for termination of RHP was unsuccessful and led to Kaveh et al.Int J Fertil Steril, Vol 11, No 4, Jan-Mar 2018              320a uterine rupture. The surgical approach is considered as the first management strategy. There are several reports of early diagnosis and laparoscopic excision of rudimentary horn (10-12). Edelman has reported a successful strate-gy treatment of RHP including the use of methotrexate (MTX) and laparoscopic excision of rudimentary horn in the first weeks of pregnancy (13). Emergency surgery after diagnosis even in cases of un-ruptured rudimentary horn has been recommended (3). Also, prophylactic re-moval of the rudimentary horn has been suggested (14). There is a report of  RHP reaching a full-term delivery that led to live birth using cesarean section (15). The re-productive outcome of a unicornuate uterus is discussed in some articles. However, afew of them have discussed the reproductive outcome after resection of rudimentary horn. Those patients who have ever undergone resection of a rudimentary horn should be considered as a high-risk group in the fallowing pregnancy (16, 17). This case re-port had no ethical consideration for patient.ConclusionRHP has high risk of death for mother, so there must be a strong clinical suspicion for the diagnosis of RHP. Although there is a major advancement in field of diag-nostic ultrasound and other imaging modalities, prenatal diagnosis has remained elusive and a laparotomy surgery is considered as a definitive diagnosis. Early diagnosis, timely resuscitation, laparotomy, and blood transfusion are the necessary management steps to save a patient.AcknowledgementsThis study was financially supported by Iran University of Medical Sciences. There is no conflict of interest in this study.Author's ContributionsM.K., A.M.K.; Contributed to conception and design. K.S.; Were responsible for overall supervision. F.F.; Draft-ed the manuscript, which was revised by M.K. All authors read and approved the final manuscript.References1. Ural SH, Artal R. Third-trimester rudimentary horn pregnancy. A case report. J Reprod Med. 1998; 43(10): 919-921.2. Scholtz M. A full-time pregnancy in a rudimentary horn of the uter-us. J Obstet Gynaecol Br Emp. 1951; 58(2): 293-296.3. Jayasinghe Y, Rane A, Stalewski H, Grover S. The presentation and early diagnosis of the rudimentary uterine horn. Obstet Gy-necol. 2005; 105(6): 1456-1467.4. Hassan CH, Karim AK, Ismail NA, Omar MH. Case report of ruptured non-communicating right rudimentary horn pregnancy: an acute emergency. Acta Medica (Hradec Kralove). 2011; 54(3): 125-126.5. Kanagal DV, Hanumanalu LC. Ruptured rudimentary horn preg-nancy at 25 weeks with previous vaginal delivery: a case report. Case Rep Obstet Gynecol. 2012; 2012: 985076.6. Fedele L, Dorta M, Vercellini P, Brioschi D, Candiani GB. Ultra-sound in the diagnosis of subclasses of unicornuate uterus. Obstet Gynecol. 1988; 71(2): 274-277.7. Bahadori F, Borna S, Behroozlak T, Hoseini S, Ayatollahi H. Failed induction in second trimester due to pregnancy in an uncommu-nicated rudimentary horn: case report. J Family Reprod Health. 2009; 3(3): 95-97.8. Tsafrir A, Rojansky N, Sela HY, Gomori JM, Nadjari M. Rudimen-tary horn pregnancy first-trimester prerupture sonographic diagno-sis and confirmation by magnetic resonance imaging. J Ultrasound Med. 2005; 24(2): 219-223.9. Samuels TA, Awonuga A. Second-trimester rudimentary uterine horn pregnancy: rupture after labor induction with misoprostol. Ob-stet Gynecol. 2005; 106(5 Pt 2): 1160-1162.10. Dicker D, Nitke S, Shoenfeld A, Fish B, Meizner I, Ben-Rafael Z. Laparoscopic management of rudimentary horn pregnancy. Hum Reprod. 1998; 13(9): 2643-2644.11. Silva PD, Welch HD. Laparoscopic removal of a symptomatic rudi-mentary uterine horn in a perimenarchal adolescent. JSLS. 2002; 6(4): 377-379.12. Yoo EH, Chun SH, Woo BH. Laparoscopic resection of a rudimentary horn pregnancy. Acta Obstet Gynecol Scand. 1999; 78(2): 167-168.13. Edelman AB, Jensen JT, Lee DM, Nichols MD. Successful medi-cal abortion of a pregnancy within a noncommunicating rudimen-tary uterine horn. Am J Obstet Gynecol. 2003; 189(3): 886-887.14. Nahum GG. Rudimentary uterine horn pregnancy. The 20th-centu-ry worldwide experience of 588 cases. J Reprod Med. 2002; 47(2): 151-163.15. Shin JW, Kim HJ. Case of live birth in a non-communicating rudimen-tary horn pregnancy. J Obstet Gynaecol Res. 2005; 31(4): 329-331.16. Okonta PI, Abedi H, Ajuyah C, Omo-Aghoja L. Pregnancy in a non-communicating rudimentary horn of a unicornuate uterus: a case report. Cases J. 2009; 2: 6624.17. Lai YJ, Lin CH, Hou WC, Hwang KS, Yu MH, Su HY. Pregnancy in a noncommunicating rudimentary horn of a unicornuate uterus: Prerupture diagnosis and management. Taiwan J Obstet Gynecol. 2016; 55(4): 604-606. Fertility Preservation in Iranian Cancer Patients

Pregnancy in non-communicating rudimentary horn of a unicornuate uterus

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Pregnancy in non-communicating rudimentary horn of a unicornuate uterus

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