Management of the uterine anomalies with the rudimentary horn. Report of two cases

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CASE REPORT

Hippokratia 2024, 28(4): 173-175

Bağlı İ, Güzel G, Ateş HE, Bozan F
Department of Obstetrics and Gynecology, University of Health Sciences, Diyarbakir Gazi Yasargil Research and Training Hospital, Diyarbakır/Turkey

Abstract

Background: Rudimentary horn (RH) uterine anomalies are rare müllerian duct development anomalies. RH pregnancies often result in rupture and may endanger the life of the mother; therefore, when RH is detected, excision of the RH is recommended.

First case: A 28-year-old G4P3 (one vaginal delivery and two cesarean sections) pregnant woman at 14 weeks gestation was admitted to the emergency department with complaints of widespread abdominal pain and fainting. The ultrasound revealed that the fetus was in the abdomen with an intact amniotic membrane, widespread free fluid, and hematoma in the abdomen, and we performed an urgent laparotomy. As a result of the rupture of the RH, massive intra-abdominal bleeding and the fetus was found to be in the abdominal cavity. We excised the ruptured RH with a right salpingectomy, and her life was saved.

Second case: A 26-year-old, G1P0, 36 weeks pregnant, underwent emergency cesarean section due to 5 cm cervical dilation and breech presentation. The left RH was detected intraoperatively and left tubal ligation was performed due to the proximity of RH to the unicornuate uterus.

Conclusion: Appropriate clinical management is crucial for patients who have RH anomalies. Excision of RH is recommended to prevent future RH pregnancy because of its high risk of rupture. Even if excision is not feasible, at least ligating the fallopian tube on the rudimentary side is an option to prevent future RH pregnancy. HIPPOKRATIA 2024, 28 (4):173-175. 

Keywords: Uterine anomalies, rudimentary horn, pregnancy, excision, fallopian tube ligation

Corresponding author: Associate Professor İhsan Bağlı, Department of Obstetrics and Gynecology, University of Health Sciences, Diyarbakir Gazi Yasargil Egitim Arastirma Hastanesi Ek Bina, Urfa Yolu, Baglar, 21090 Diyarbakir, Turkey, tel: +905557123342, e-mail: ihsanbagli@gmail.com

Introduction

The unicornuate uterus with rudimentary horn (RH) uterine anomalies is a rare anomaly of the müllerian duct development. The unicornuate uterus is found in 0.4 % of all women, and in approximately 84 %, the unicornuate uterus has a rudimentary horn on the other side1. Incidence of rudimentary horn pregnancy (RHP) is even more rare, ranging from 1 in 76,000 to 1 in 150,000 pregnancies2. RHP may lead to rupture of the horn by the second trimester in 80-90 % of all cases3. Removal of the RH along with the ipsilateral fallopian tube has been recommended for the prevention of RHP and chronic pelvic pain3-5. Despite this uterine anomaly, which can threaten the life of the mother, few case reports and one of the cases currently reported show that some patients did not undergo excisional surgery or the diagnosis was missed, although it could have been diagnosed incidentally in previous surgeries6-8. Herein, we present our approach in two cases: unicornuate uterus and rudimentary horn anomaly. 

First case description

A 28-year-old G4P3 (one vaginal delivery and two cesarean sections) pregnant woman at 14 weeks gestation was admitted to the emergency department with complaints of widespread abdominal pain and fainting in June 2024. According to the ultrasonographic findings performed in a private hospital two days before her admission, she was told that there might be a pelvic mass. The anamnesis did not include any abnormality recording of her reproductive system despite two previous cesarean sections. The ultrasound performed on admission revealed a live 14-week fetus in the abdominal cavity with an intact amniotic membrane, widespread free fluid, and hematoma in the abdomen. Under general anesthesia, we performed an urgent laparotomy via a previous Pfannenstiel cesarean section incision with a diagnosis of uterine rupture. Intraoperatively, we encountered a rupture of the non-communicating RH with a unicornuate uterus, massive intra-abdominal bleeding, and the fetus was found to be in the abdominal cavity (Figure 1). Adhesions between the unicornuate uterus, omentum, and abdominopelvic wall structures were released. The ruptured RH was excised with ipsilateral salpingectomy, and the patient’s life was saved. A Foley catheter was placed into the abdomen for fluid drainage. As a result of complete blood count taken before surgery, hemoglobin was found to be 8.2 mg/dl. Blood products were not given to the patient. The patient was discharged uneventfully on the second day of the hospitalization.

Figure 1: Intraoperative view of ruptured non-communicating rudimentary horn pregnancy with unicornuate uterus at emergent laparotomy.

Second case description

A 26-year-old, G1P0, 36 weeks pregnant, underwent emergency cesarean section under regional anesthesia via Pfannenstiel incision due to 5 cm cervical dilation and breech presentation in May 2023. A 2,600 gr baby boy was born with an 8-9 Apgar score. A left RH was detected intraoperatively. No cavitary connection was detected between the RH and the unicornuate uterus. Thanks to regional anesthesia, this uterine abnormality was discussed with the patient intraoperatively for prevention of possible future pregnancy in the RH. After informed consent, left tubal ligation was performed without excision of RH due to the near connection of RH muscularity to the unicornuate uterus (Figure 2). This uterine abnormality was noted in the patient’s operation folder as a unicornuate uterus with non-communicating RH. The patient was discharged uneventfully on the second day of the cesarean section.

Figure 2: Intraoperative view of incidentally detected noncommunicating rudimentary horn with unicornuate uterus and ligation of the ipsilateral fallopian tube during cesarean section.

Discussion

Early diagnosis is important to avoid life‑threatening complications such as a rupture of the RHP and long‑term morbidity in terms of debilitating chronic pelvic pain5,8,9. As stated in the American Society of Reproductive Medicine Guideline in 2021, these müllerian anomalies consist of a wide variety of types of structural anomalies10. According to Cobec et al and Kozar et al there are often difficulties in diagnosing these uterine abnormalities which have a wide variety of types, and difficulties in diagnosis increase during the further gestational weeks9,11. In the first reported case, the evaluation of the uterine anomaly as a pelvic mass in the ultrasound report two days before the rupture was most likely due to the advanced gestational age. For example, Kozar et al reported a patient with RHP, but they interpreted it as a bicornuate uterus during the first step of the evaluation9. We primarily use ultrasound to evaluate the pelvic organs, but when a uterine anomaly is suspected, the best diagnostic tool is magnetic resonance imaging10. Although RH is recommended to be excised because it is known to cause rupture of the horn in the second trimester in 80-90 % of all cases3, it was noticed that RH excision was not performed during the two previous cesarean sections in the first reported case. Singh et al reported three missed-diagnosis patients, two of whom had one and two cesarean sections, respectively, and one who had gynecological laparoscopic surgery and was subsequently diagnosed with RH8, and as they stated in their report, these missed diagnoses are similar to the first reported case. Kaveh et al also presented a patient who went into hypovolemic shock due to a ruptured RHP and had a previous cesarean section history7. Furthermore, in one case of non-ruptured RHP, the patient stated that she had had a previous cesarean section and did not know that she had a uterine abnormality6. Interestingly, this non-ruptured RHP was terminated laparoscopically, and the rudimentary horn was repaired by Ma et al6 These missed diagnoses may be due to some surgeons suturing the uterus intra-abdominally without exteriorization of the uterus during cesarean section12. This method may be an obstacle to precisely exploring the uterus and adnexa. Another reason surgical intervention was not performed for the RH in the previous cesarean sections of these cases (our first case and the others mentioned in the discussion) may be that the surgeons avoided additional surgery. Even if no surgical intervention is performed, the patients should be informed of the presence of these anomalies after cesarean section. In the second reported case, the excision of the RH might have been difficult due to being very close to the unicornuate uterus. Therefore, the patient was informed intraoperatively about the uterine anomaly, and at least the option of tubal ligation of the RH side was offered and performed. Pregnancy in a non-communicating RH can be explained by the theory of the transmigration of sperm from the contralateral fallopian tube13. The ligation of the RH side fallopian tube can blockage this transmigration

In conclusion, appropriate clinical management is crucial for patients who have RH anomalies. Excision of RH is recommended to prevent future RH pregnancy because of its high risk of rupture. When encountered with some surgical difficulties, at least ipsilateral ligation or excision of the fallopian tube on the rudimentary side is an option to prevent future RHP.

Conflicts of interest

The authors declare no conflicts of interest.

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