Salpingo-oophorectomy for twisted ovarian dysgerminoma in a prestigious pregnancy: challenges and outcomes. A case report

Abstract

Introduction:

Cancer during pregnancy is uncommon. Symptoms caused by ovarian dysgerminoma in pregnancy can be difficult to recognize, leading to a delay in diagnosis and treatment. In addition, treatment of twisted ovarian dysgerminoma in pregnancy to improve the maternal and perinatal outcomes is the main challenges that confront obstetricians and gynecologic oncologists because surgery often results in preterm labor and delivery.

Case presentation:

A 29-year-old woman primigravida came with severe lower abdominal pain. An intrauterine foetus at 19 weeks of gestation and an enlarged left ovarian mass measuring 15.3 × 10.3 × 8 cm were discovered by pelvic ultrasonography. A left salpingo-oophorectomy was performed, and the histology revealed an ovarian dysgerminoma. She had a spontaneous vaginal delivery at term.

Discussion:

Dysgerminoma is the most frequent malignant ovarian germ cell tumor in females. Due to physiological changes, it may be a challenge to diagnose ovarian torsion in early pregnancy. In addition, the treatment of large ovarian tumors in pregnancy is a challenge because surgery often results in preterm labor and delivery. Fertility-sparing surgery is acceptable in young women. Chemotherapy for ovarian dysgerminoma is recommended for International Federation of Gynecology and Obstetrics (FIGO) stages II, III, and IV.

Conclusion:

There are many challenges when performing salpingo-oophorectomy for a large twisted ovarian tumor in early pregnancy. The decision must balance the risks of surgery against the possibility of losing the pregnancy and the urgency of acting to protect the mother’s health. The management depends on the gestational age, tumor stage, and the patient’s desire to continue the pregnancy.

Introduction

Dysgerminoma is the most frequent malignant ovarian germ cell tumor in females, accounting for about 2%. Cancer during pregnancy is uncommon. Ovarian dysgerminoma accounts for about 0.0002–0.001%^[1,2]. The ovarian vascular pedicle may rotate completely or partially, causing ovarian torsion, which blocks arterial and venous influx and outflow^[3]. Timely antenatal diagnosis and treatment of the large ovarian dysgerminoma to improve the maternal and perinatal outcomes are the main challenges that confront obstetricians and gynecologic oncologists^[4]. Women who are pregnant account for 13.7% of all tubo-ovarian torsion occurrences. Frequently, adnexal torsion happens in the first and early second trimesters of pregnancy^[5]. Due to physiological changes, it may be difficult to diagnose ovarian torsion in the early stages of pregnancy. Abdominal pain and nonspecific symptoms are common symptoms. Missed diagnosis of ovarian torsion can result in ovarian necrosis, sepsis, and threaten the pregnancy^[6]. Surgical intervention, especially in the early stage of ovarian malignancy, is the gold standard for diagnosis and treatment^[3]. The objective of this article is to provide a comprehensive understanding of the management of twisted ovarian dysgerminoma in early pregnancy and highlight the importance of timely intervention, particularly in cases of malignancy. This case report is presented in line with the SCARE criteria^[7].

HIGHLIGHTS

• Timely antenatal diagnosis and treatment of the large ovarian tumor to improve the maternal and perinatal outcomes are the main challenges that confront obstetricians and gynecologic oncologists.

• Symptoms caused by ovarian dysgerminoma in pregnancy can be difficult to recognize, leading to a delay in diagnosis and treatment.

• Fertility-sparing surgery is acceptable in young women. Chemotherapy for ovarian dysgerminoma is recommended for FIGO stages II, III, and IV.

Case report

A 29-year-old woman primigravida, with a history of primary infertility for 9 years (managed out of our facility), reported having sudden and severe lower abdominal pain for the past day. The abdomen was gravid with positive rebound on palpation. She denied, however, having experienced fever, vomiting, shoulder pain or vaginal leakage, spotting or bleeding. No urinary symptoms. The internal pelvic examination was unremarkable. The tumor markers serum β-HCG and LDH were elevated. However, the other laboratory investigations (FBC, LFT, RFT, electrolytes, AFP, CA 125, CEA, PT, and PTT) were within the normal range. The pelvic ultrasound showed a gravid uterus with a live intrauterine fetus corresponding to 19 weeks of gestation and an anteriorly fundal placenta. An enlarged left ovarian mass measuring 15.3 × 10.3 × 8 cm with well-defined borders, lobulated contours, and component lobules, with heterogeneous echogenicity, was discovered. The right ovary was measuring 6.3 × 3.1 × 2 cm. The above features gave the impression of a huge twisted left ovarian mass in pregnancy at 19 weeks of gestation. Under general anesthesia, the patient benefited from an emergency exploratory laparotomy, which revealed a gravid uterus with a large left ovarian mass, FIGO stage IA (Fig. 1). The right ovary appeared normal. No ascites was found in the abdominal cavity. The wall of the abdominal cavity, the diaphragm and the omentum were free of any lesions. Left salpingo-oophorectomy was performed (Fig. 2). The histopathology revealed ovarian dysgerminoma. Pregnancy was maintained with aggressive postoperative luteal support (progesterone pessary) for 1 month, anti-infective treatment for 7 days (Ceftriaxone and Metronidazole), and analgesics for pain management. Close monitoring of the rest of the pregnancy period was done with an ultrasound 2 days after the surgery, then every 4 weeks, and it was uneventful. She was readmitted due to preterm labor at 30 weeks of gestation, which was due to a UTI during pregnancy. She was given IV Ceftriaxone 1 g odx2/7 then Ampiclox 500 mg tds ×5/7, Paracetamol tabs 1 g tds for 7 days, Magnesium sulphate as a tocolytic and neuroprotection 1 g/h for 24 h, and Dexamethasone 6 mg bid ×2/7 for fetal lung maturation. The uterine contractions subsided, and the patient was discharged after 3 days. She was followed up, and her pregnancy continued unremarkably. She had a spontaneous vaginal delivery at 38 weeks of gestation, giving a healthy male baby, Apgar 8-9-10, birth weight 2860 g. The postpartum period was uneventful. This case report is presented in line with the SCARE criteria^[7].

Figure 1.

Left twisted ovarian dysgerminoma in pregnancy at 19 weeks of gestation.

Figure 2.

Left salpingo-oophorectomy due to twisted ovarian dysgerminoma in pregnancy at 19 weeks of gestation.

Discussion

Dysgerminoma is the most frequent malignant ovarian germ cell tumor in females, accounting for about 2%. In pregnant women, ovarian dysgerminoma is very rare, accounting for about 0.0002–0.001%. It usually has a unilateral onset in 85% of cases and is diagnosed at an early stage^[1]. Ovarian dysgerminoma can occur in women aged from 7 months to 70 years. It predominantly affects women aged 20–30 years old. However, it is commonly detected in adolescent women, and may present bilaterally in 15% of cases and tends to grow rapidly, facilitating early diagnosis. Clinical complications such as rupture, hemoperitoneum, or torsion can cause acute abdominal presentations, necessitating urgent intervention^[1,8,9].

One of the major gynecologic emergencies among women of reproductive age is ovarian torsion. Most ovarian tumor torsion happens during the reproductive age. More pregnant women (22.7%) than nonpregnant women (6.1%) present with ovarian torsion^[6,10,11]. In our instance, the patient was a 29-year-old woman, primigravida with a history of primary infertility. She experienced an ovarian tumor torsion at 19 weeks of gestational age.

Cancer during pregnancy is uncommon. Symptoms of ovarian dysgerminoma in pregnancy can be difficult to recognize, leading to a delay in diagnosis and treatment. In addition, the diagnosis of ovarian tumor torsion in pregnancy may be a challenging one since palpation and ultrasound imaging of the ovaries are more difficult than in nonpregnant patients. Furthermore, the symptoms of ovarian torsion, such as nausea, vomiting, and abdominal pain, are nonspecific and can resemble those of several obstetric and surgical disorders, including ovarian hyperstimulation, premature labor, acute appendicitis, and pyelonephritis; the diagnosis may be missed^[2,12]. In our case, the patient had been experiencing severe lower abdominal pain for the past day without any other associated symptoms. The ultrasound confirmed the twisted adnexal mass, and the histology results confirmed the diagnosis of ovarian dysgerminoma.

The treatment of huge ovarian tumors generally involves surgical removal of the tumor. While laparoscopic management is currently considered the gold standard for adnexal tumors, many authors consider tumor dimensions exceeding 8–10 cm as a technically challenging factor for this approach. In addition, large ovarian tumors are difficult to treat during pregnancy because surgery often results in preterm labor and delivery. Therefore, the decision must balance the risks of surgery against the possibility of losing the pregnancy and the urgency of acting to protect the mother’s health, particularly in a highly needed pregnancy, and or a patient who desires to continue the pregnancy. The objective of surgery is to treat the twisted ovarian dysgerminoma while taking the pregnancy’s viability into account.

An oophorectomy, which removes the ovary that contains the corpus luteum, may result in miscarriage in the early stages of pregnancy. Literature states that surgical intervention done even in the second trimester may result in miscarriage, intrauterine growth restriction, and preterm labor and delivery^[13,14]. In our case, the patient had preterm labor at 30 weeks of gestation that was managed successfully. She delivered vaginally at term, an alive male baby, Apgar 8-9-10, birth weight 2860 g. The postpartum period was unremarkable. The patient was counselled and planned for surveillance every 3 months for 2 years, including physical examination, serum β-HCG, LDH, and pelvic ultrasound every 6 months.

Unilateral salpingo-oophorectomy, which was performed in our case, is the recommended fertility-sparing surgical treatment, especially for early stages of ovarian dysgerminoma. The prognosis is favorable, with nearly a 100% 5-year survival rate. There are several studies which reported that even for advanced stages of ovarian dysgerminoma, fertility-sparing surgery is an acceptable treatment, especially for young women still desirous of pregnancy, since the overall survival rate is high and the recurrence rate is low. Following the surgery, adjuvant chemotherapy must be instituted^[15].

Chemotherapy for ovarian dysgerminoma is recommended for FIGO stages II, III, and IV. Chemotherapy is recommended based on pathological evidence, especially in advanced-stage, mixed epithelial and germ cell tumors, large tumor size, and rapidly increasing ascites^[1,9,16]. In our case, the patient underwent fertility-sparing surgery without chemotherapy because of the stage of the disease, FIGO stage IA.

In the literature, Awada et al reported a case of rapidly growing ovarian dysgerminoma in a 28-year-old pregnant woman. The patient was delivered by cesarean section at 37 weeks weeks, a healthy female fetus weighing 2500 g. She also underwent fertility-preserving surgery of the left adnexa followed by chemotherapy. No abnormalities were reported 2 years after the surgery^[9]. Zhang et al reported a case of pregnancy with a giant ovarian dysgerminoma stage II B. The patient was delivered by cesarean section at 38 weeks of gestation due to severe abdominal pain (alive baby 2540 g Apgar score of 9 and 10) and underwent fertility-preserving surgery. However, the patient refused chemotherapy postoperatively, and no tumor recurrence was observed after 42 days, 3 months, and 6 months after the surgery^[1]. Sumedha G. et.al report a case of twisted ovarian dysgerminoma stage III with omental metastasis in an 11-year-old female who came with an abdominal mass and torsion. The patient underwent fertility-sparing surgery followed by adjuvant chemotherapy^[8].

In our case, the patient underwent a fertility-sparing surgery without chemotherapy due to her FIGO stage IA with no recurrence observed from 19 weeks of gestation to 42 days postpartum and had a spontaneous vaginal delivery at 38 weeks of gestation, giving a healthy male baby, Apgar 8-9-10, birth weight 2860 g. We succeeded in the spontaneous vaginal delivery, which is the preferred mode rather than caesarean section. The benefits of spontaneous delivery are less blood loss, less operative risk, and lower infection risk with shorter length of stay at the hospital. However, the difference in the mode of delivery in our case and the literature may be justified by the difference in gestational age at the time of diagnosis and treatment.

Conclusion

There are many challenges when performing a salpingo-oophorectomy for a large twisted ovarian tumor in early pregnancy. The decision must balance the risks of surgery against the possibility of losing the pregnancy and the urgency of acting to protect the mother’s health. The management depends on the gestational age, tumor stage, and the patient’s desire to continue the pregnancy.

Ethical approval

Not applicable.

Consent

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

Sources of funding

None.

Author contributions

M.M.C.: received and examined the patient, and wrote the manuscript. D.B.: prepared the images and read the main manuscript. B.M.: performed the surgical intervention, read, and corrected the manuscript.

Conflicts of interest disclosure

All the authors have any possible conflicts of interest.

Guarantor

Mpoyi Mukeba Constantin.

Research registration unique identifying number (UIN)

(1) Name of the registry: Medical Records of Muhimbili National Hospital. (2) Unique identifying number or registration ID: M88-33-21. (3) Hyperlink to your specific registration (must be publicly accessible and will be checked): https://www.mnh.or.tz/index.php/directories/information-andcommunication-technology/medical-records-department.

Acknowledgements

Mpoyi Mukeba Constantin acknowledges with gratitude the support from the Else-Kroner-Fresenius Stiftung foundation through the Bourse d’Excellence Bringmann aux Universités Congolaises, BEBUC Scholarship program. Funds received were used outside of this study.

Data availability statement

A copy of the written consent is available for review by the editor-in-chief of this journal on request. The data file is available in the medical record system of Muhimbili National Hospital.