Spontaneous uterine rupture as a life-threatening presentation of molar pregnancy: a case report

Abstract

Introduction

Gestational trophoblastic disease is the term used to describe the heterogeneous group of interrelated lesions that arise from abnormal proliferation of placental trophoblasts. The clinical presentations of gestational trophoblastic tumor are vaginal bleeding, uterine enlargement greater than expected for gestational dates, hyperemesis, secondary clinical hyperthyroidism, and less common presentations such as pregnancy-induced hypertension in the first or second trimester and theca lutea cyst torsion. It is a rare incident to find uterine rupture due to a molar pregnancy. Most uterine ruptures reported so far were all malignant histologic types of the spectrum.

Case presentation

We present a case of ruptured uterus as a complication of complete molar pregnancy presenting with shock and severe anemia. A 37-year-old Ethiopian gravida 6 para 5 with 5-month amenorrhea came with vaginal bleeding, abdominal pain, and symptoms of anemia. Her hemoglobin was 2.8 g/dl, and there was a honeycomb-appearing endometrial mass and free fluid in the general peritoneum. She underwent emergency laparotomy, where a hysterectomy was done for cornual uterine rupture. She was followed with serial serum human chorionic gonadotropin and declared cured after 6 months of surveillance.

Conclusion

Even though it is rare, a ruptured uterus in a molar pregnancy can be a catastrophic complication, presenting with massive hemoperitoneum and hemorrhagic shock. Hysterectomy, along with vascular filling with crystalloid and transfusion of blood products, can save a patient’s life. Patients can be followed with serial serum human chorionic gonadotropin for any transformation to gestational trophoblastic neoplasia.

Introduction

Uterine rupture, which occurs almost always in the gravid uterus, refers to the complete disruption of the uterine layers. The most common causes of uterine rupture are neglected obstructed labor and scarred uterus in labor [1, 2]. Both uterine rupture and perforation, which indicate noniatrogenic and iatrogenic uterine wall injury, respectively, are linked to significant morbidity and occasionally death [3].

Gestational trophoblastic disease encompasses a spectrum of pregnancy-related disorders, consisting of the premalignant disorders of complete and partial hydatidiform mole and the malignant gestational trophoblastic neoplasia (GTN), which includes invasive mole, choriocarcinoma, epithelioid trophoblastic tumor, and the rare placental-site trophoblastic tumor [4, 5].

The clinical presentations of gestational trophoblastic tumor are vaginal bleeding (80–90%), uterine enlargement greater than expected for gestational dates (28%), hyperemesis (8%), secondary clinical hyperthyroidism (2%), and less common presentations such as pregnancy-induced hypertension in the first or second trimester and theca lutea cyst torsion [6–8]. Gestational trophoblastic neoplasia, which consists of the malignant end of the spectrum, can present with metastasis-related emergency presentations such as symptoms of lung involvement (shortness of breath, hemoptysis, cough, and chest pain), neurologic manifestations from brain metastasis (seizure, deficit, and headache), and abdominal pain (liver or gastrointestinal involvement); unusual locations, such as ischemia of lower extremity due to femoral artery metastasis, gingival metastasis, and retroperitoneal, have also been reported [5, 9–11].

It is a rare incident to find uterine rupture due to a molar pregnancy. Most uterine ruptures reported so far were all malignant histologic types of the spectrum [9, 12].

We herein present the case of a patient managed for hemorrhagic shock secondary to a uterine rupture due to gestational trophoblastic disease (GTD), which is a complete mole. This case report has been reported in line with the CARE guidelines [13].

Case presentation

This case describes a 37-year-old Ethiopian gravida 6 para 5 mother who claims 5-month amenorrhea presented to the gynecologic emergency unit with vaginal bleeding of 5 days duration, which was intermittent and associated with passage of grape-like tissue. Associated with this, she also has a history of abdominal pain, easy fatiguability, shortness of breath, palpitation, and nonprojectile vomiting of ingested matter of 2 days duration. She denied having any surgical treatment or trauma to the abdomen. One day before her current admission, she was evaluated at a local clinic for vaginal bleeding and referred for higher-level care, but due to financial reasons, she was delayed. Her condition subsequently deteriorated, necessitating emergency ambulance transfer to this hospital.

All her previous pregnancies were vaginal deliveries, and her youngest child is 3 years old. The patient denied any personal history of chronic conditions or allergies, as well as any known heritable disorders in her first-degree relatives.

On physical examination, the patient appeared acutely ill, exhibiting signs of distress due to pain. Her vital signs were as follows: blood pressure of 80/50 mmHg, pulse rate of 132 beats per minute, respiratory rate of 22 breaths per minute, and temperature of 37.1 °C.

Additionally, her conjunctiva appeared pale. Abdominal assessment showed a distended abdomen and mild uterine tenderness, and the uterus measures 22 weeks, with soft and boggy consistency. There was positive shifting dullness and fluid thrill. Pelvic examination showed a bulged cul-de-sac, the parametrium felt free, and no adnexal mass was palpable.

Her laboratory results revealed that her serum level of human chorionic gonadotropin (HCG) was 242,000 IU/L, her white blood cells were 10.3 × 10⁹/L, her hemoglobin was 2.8 g/dL, and her platelets were 196 × 10⁹/L. A bedside ultrasound was done and revealed with intrauterine content of 18 cm × 0 cm, which was a honeycombed heterogeneous mass with multiple small anechoic spaces filling the uterine cavity, and there was free fluid in the general peritoneum, as displayed in Fig. 1.

Fig. 1.

Transabdominal sonography showing an intrauterine mass with “snowstorm” appearance (arrowhead) and free fluid in the general peritoneum (arrow)

She consented to an urgent laparotomy after discussing the plan of surgery, including the possibility of hysterectomy, and was wheeled to the theater immediately. Intraoperatively, a fundal uterine rupture of approximately 3 cm was found with a hemoperitoneum of approximately 3000 mL. There was expulsion of clustered friable cystic endometrial content into the peritoneal cavity, as shown in Fig. 2. The tubes and ovaries appeared normal bilaterally. Total abdominal hysterectomy and abdominal washout were done with normal saline, and the abdomen was closed in layers. She was transfused with two units of blood intraoperatively and two units postoperatively, and her hemoglobin levels increased. There were no intraoperative complications. She recovered well and was discharged on the third postoperative day with analgesia and a therapeutic dose of iron sulfate.

Fig. 2.

Intraoperative finding of a ruptured uterus around the left cornua, measuring around 3 cm (arrow), and with the expulsion of gestational trophoblastic disease products with a healthy-looking ovary (O) and fallopian tube (T)

Histopathologic examination of the findings is consistent with a complete mole and lacks invasion of the myometrium. The patient then followed up every 2 weeks for 1 month and monthly for a total of 6 months by serial serum human chorionic gonadotropin (HCG) and was declared cured after 3 months of an undetectable level of HCG.

Discussion

According to the 2020 World Health Organization (WHO) Classification of Female Genital Tumors, they can be categorized into molar pregnancies/hydatidiform moles, gestational trophoblastic neoplasms, tumor-like (non-neoplastic) lesions, and abnormal (nonmolar) villous lesions [14].

The most prevalent type of GTD is the hydatidiform mole (molar pregnancy), which has an abnormal placental derivation that is characterized by an excess of placental chorionic tissue. Geographical differences exist in the incidence of molar pregnancy. Based on genetics, visual and microscopic features, and clinical characteristics, it is further divided into three categories: partial, complete, and invasive moles [5, 15]. Risk factors include age extremes, previous history of GTD, and race.⁵

Given that every normal pregnancy includes a physiological invasion phase during placentation, it is quite likely that a myometrial invasion occurs in the majority of GTDs. However, it might be challenging to determine whether an aberrant myometrial invasion is present using pelvic ultrasonography or suction curettage, the first-line treatment for moles [12, 15].

Clinical diagnosis of uterine rupture can be difficult as it mimics different causes of acute abdomen. Ranging from ruptured ovarian cyst, which is the most common cause of nontraumatic hemoperitoneum in women, to rare causes of rupture of pregnancy in uterine anomaly, the differential diagnosis is diverse [16, 17]. Ultrasound (US) is the initial imaging modality of choice, as it may be able to depict the site of the rupture and the level of hemoperitoneum [3, 18]. Computed tomography (CT) can be helpful when ultrasound is inconclusive or if need for detailed pelvic pathology in cases of metastasis suspicion. Magnetic resonance imaging (MRI) and catheter angiography can be used for more detailed information, such as the size of the rupture and the presence of active bleeding. Catheter angiography can also be therapeutic in the selective embolization of the uterine artery [3].

Management of a ruptured uterus with intrauterine GTD depends on the clinical condition of the patient, future fertility needs, and the technical feasibility of repairing the ruptured site. Most reported cases have been managed so far with hysterectomy with subsequent chemotherapy, as most of the histologic types were malignant end of the spectrum [12, 19, 20]. There is a report of a successful pregnancy after cornuectomy followed by chemotherapy for uterine rupture due to GTN [21]. Laparoscopic excision of an invasive mole followed by chemotherapy has also been reported [22]. Surveillance by serial serum HCG is required regardless of the mode of initial management, either evacuation or hysterectomy. Most agree on every 1–2 week follow-up until normalization and monthly for 6 months for complete molar pregnancy [23, 24].

Conclusion

Even though it is rare, a ruptured uterus in a molar pregnancy can be a catastrophic complication, presenting with massive hemoperitoneum and hemorrhagic shock. Hemoperitoneum may be overlooked as a reason for hemorrhagic shock when patients present with simultaneous vaginal bleeding.

Hysterectomy, along with vascular filling with crystalloid and transfusion of blood products, can save a patient’s life. Patients can be followed with serial serum HCG for any transformation to GTN.

Author contributions

EFD: conceptualization, data curation, postoperative follow-up of the patient, conceptualization, writing the original manuscript, data curation, and review editing. AAD: patient diagnosis, operation, supervision, validation, and final manuscript approval. SFG: conceptualization, data curation, review editing, and final manuscript approval. SEK: operation, conceptualization, data curation, investigation, and final manuscript approval. GSW: data curation, investigation, and final manuscript approval. ZDA: data curation, investigation, and final manuscript approval.

Funding

No funding received.

Data availability

The data supporting this case report are not publicly available to protect patient privacy but are available from the corresponding author upon reasonable request.

Declarations

Ethics approval and consent to participate

Formal written informed consent for the case and pictures for intended educational purposes and the patient’s identifying information has been anonymized for privacy and confidentiality, and it will be available upon request. Institutional review board approval is not applicable to this case report.

Consent for publication

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Competing interests

The authors declare no competing interests in computation.