- Adnan Incebiyik#dr dot aincebiyik at gmail dot comHarran University, School of Medicine, Department of Obstetrics and Gynecology, Sanlıurfa, Turkey
- Nese Gul HilaliHarran University, School of Medicine, Department of Obstetrics and Gynecology, Sanlıurfa, Turkey
- Aysun CamuzcuogluHarran University, School of Medicine, Department of Obstetrics and Gynecology, Sanlıurfa, Turkey
- Hakan CamuzcuogluHarran University, School of Medicine, Department of Obstetrics and Gynecology, Sanlıurfa, Turkey
- Ahmet SekerHarran University, School of Medicine, Department of General Surgery, Sanlıurfa, Turkey
- Mehmet VuralMarmara School of Medicine, Department of Obstetrics and Gynecology, Istanbul, Turkey
Choriocarcinoma is a rare neoplasia characterized by distant metastasis and the invasion of trophoblastic tissue into the myometrium. The problems that take place during diagnosis, treatment and follow-up of the disease may lead to life-threatening complications, such as uterine rupture. Here we present a case of a patient who was previously operated on with vacuum aspiration for molar pregnancy, who did not attend the follow-up examinations and who was finally admitted with acute abdomen due to choriocarcinoma-caused uterine rupture.
Choriocarcinoma originates from trophoblasts and has the tendency to metastasize early. Although the exact cause is unknown, it is assumed to have a genetic background. The sole presence of paternal chromosomes in conceptus is believed to lead to the hyperproliferation of trophoblasts and subsequently to the progression of choriocarcinoma (1). It has an incidence of 1/50,000 and presents itself with abnormal postpartum vaginal bleeding. Even though it is rare, acute abdomen due to hemoperitoneuma as a result of uterine perforation may be the first symptom of choriocarcinoma (2,3).
Here we evaluated a case of uterine rupture due to choriocarcinoma. The patient was operated on with vacuum aspiration molar pregnancy 4.5 months prior and did not attend the follow-up examinations. Thus, choriocarcinoma could not be detected in the early phase and eventually led to the rupture of the uterus.
A 45-year-old patient was admitted to our hospital with vaginal bleeding for two days and increasing abdominal pain, nausea and vomiting for the previous 10 hours. The patient had a history of nine normal births and three abortions. Approximately 4.5 months earlier, the patient was operated on with vacuum aspiration for molar pregnancy, but then did not attend the follow-up examinations. The patient had no symptoms until two days prior to admission to the hospital.
During the physical examination, the patient was pale, afebrile and tachycardic (pulse: 110 bpm) and had a blood pressure of 70/40 mm Hg and sensitivity in the whole abdomen, especially in the lower part. A small amount of uterine bleeding from the cervix was detected in the speculum examination, and the uterus was enlarged during the bimanual examination. During the abdominal ultrasonography, the uterus was larger than normal (25x35 mm and 55x40 mm) and irregular. A heterogeneous mass was localized in the fundus, and a high amount of intra-abdominal free fluid was present. The loss of serosal tissue continuity on the mass was interpreted as a sign of uterine perforation. There were no abnormalities in laboratory tests except for haemoglobin (7.7 mg/dl), hematocrit (26.1%) and leukocyte (20.360/mm³) levels.
The decision was made to perform an emergency laparotomy on the patient. Approximately 1,500 ml of blood and clot was drained from the abdomen during the operation. During the subsequent examination, the uterus was found to be enlarged, and an actively bleeding lesion (approximately 60x40 mm and 30x40 mm) was detected in the fundus (Figure). A total abdominal hysterectomy was performed, as the patient was 45 years old and did not desire to conceive. Two units of erythrocyte suspensions were administered operatively. During the examination of the hysterectomy specimen in the operating room, it was observed that the tumour tissue had invaded the full layer of the uterus wall.
The β-human chorionic gonadotropin (β-hCG) level was 1.012.027 mIU/ml in the preoperative blood sample. Thus, together with the histopathological diagnosis of the surgical specimen, the underlying cause of the rupture was identified as choriocarcinoma.
Thoracic tomography was taken on postoperative day 2 to detect possible metastases, and an appearance compatible with metastasis was detected in both posterolateral segments of the lower lung lobes. There were no abnormalities in the entire abdominal and cranial magnetic resonance images.
On postoperative day 8, the case was monitored by the Department of Medical Oncology to evaluate the chemotherapy, and multiagent chemotherapy (EMA-CO) treatment was started due to the presence of stage 3, high-risk conditions. After the fourth cycle of chemotherapy, the β-hCG level was <5 mIU/ml, and the patient was administered with two additional cycles of chemotherapy. There was no sign of recurrence in the 12th-month follow-up examination of the patient.
In our case presentation, we emphasize that uterine rupture secondary to choriocarcinoma may be the underlying cause of acute abdomen due to hemoperitoneum in patients in the reproductive age group with a recent history of pregnancy.
Choriocarcinoma is a rare neoplasia characterized by the invasion of trophoblastic tissue into the myometrium and distant metastasis. The condition generally presents itself with localized bleeding, mostly with postpartum vaginal bleeding. Less frequent symptoms include haemoptysis, cerebral bleeding, metastatic bleeding (e.g. liver hematoma) and acute abdomen secondary to hemiperitoneum and uterine rupture. There are only eleven reports on uterine rupture owing to choriocarcinoma in the literature (April 2014, Mesh database, Keywords: choriocarcinoma and uterine rupture). Although the exact cause of uterine rupture in choriocarcinoma is not known, the following theories have been proposed to explain this condition. Trophoblasts can invade the uterine veins, and blood vessel may be damaged. As a result of such damage, multiple infarctions occur due to thrombosis, vascular aneurysms and intratumoral bleeding (3). In line with its hypervascular nature, myometrial invasion (4) – and depending on the chemotherapy regimen used – rapid trophoblast death can lead to necrosis and eventually uterine perforation (5). The presence of a 90% necrotic choriocarcinoma mass led us to speculate that necrosis, in addition to tumoral invasion, may be the underlying cause of perforation in our case.
A high level of bleeding occurred due to uterine rupture, and the patient was admitted to the hospital with an acute abdomen. Typically, ectopic pregnancy with hemoperitoneum, the rupture of a corpus luteum cyst and adnexal torsion should be considered during the differential diagnosis. Ultrasonography and β-hCG levels may aid in the diagnosis. In the ultrasonography, an irregular, heterogeneous mass in the fundus, hemoperitoneum, and the loss of serosal tissue continuity on the mass were detected, and this was interpreted as uterine perforation; other potential causes were disregarded.
Patients with acute abdomen from uterine perforation and choriocarcinoma should be treated for hypovolemia and anaemia. In addition, the bleeding foci should be stopped with a surgical procedure. A total abdominal hysterectomy is the preferred treatment method in women who do not desire to conceive and who are operated on with explorative laparotomy under emergency conditions due to severe active intra-abdominal bleeding. However, conservative surgery can be employed in women who wish to preserve their fertility and have lesions that are limited to the uterus (5). In our case, we also planned a hysterectomy to remove the bleeding focus. The patient was operated on under emergency conditions, and the transfusion of only two units of erythrocyte suspension was sufficient to maintain hemodynamic stability.
Owing to the high vascular invasiveness of the tumour tissue, local aggressive spreading and the risk of early systemic metastasis was high. The most common site of metastasis is the lungs and then the vagina, brain, liver, and kidneys, respectively (6). The lungs, which are the most frequently metastasized organ in choriocarcinoma, were also observed in our patient through imaging techniques. Metastatic foci were not observed in the thoracic tomography after six cycles of chemotherapy. In our case, the local invasion of the tumour tissue was the underlying cause for uterine perforation.
Many institutions use the International of Obstetrics and Gynecology (FIGO) grading and scoring system to determine the suitable treatment for choriocarcinoma cases and to illustrate diffuse of the disease. Single-agent chemotherapy is used in non-metastatic and low-risk choriocarcinomas (score ≤6), whereas multi-agent chemotherapy and radiotherapy are used for high-risk, metastatic choriocarcinomas (score >6) (2,7,8). Our case was identified as stage 3 due to the presence of lung metastasis; the total score was identified as 8 according to the risk scoring system. Therefore, we administered multiagent chemotherapy because of the presence of a high-risk metastatic condition.
Serial β-hCG measurements are employed to monitor patients administered with chemotherapy and to identify the success rate of the treatment. Since β-hCG is secreted from viable trophoblasts, serum β-hCG levels are expected to decrease with a decreasing tumour burden. Chemotherapy is continued until β-hCG levels return to normal, and two or three cycles of chemotherapy are recommended to eradicate all living tumour cells (9). In our case, there was a 34% reduction in β-hCG levels following the hysterectomy (pre-op: 1.012.027 mIU/ml; post-op: 344.132 mIU/ml). During the fourth cycle of EMA/CO protocol, which was administered as a cytotoxic treatment, negative β-hCG levels were detected, and an additional two cycles of chemotherapy were administered.
In conclusion, uterine rupture due to choriocarcinoma is a life-threatening condition and should be considered among the differential diagnoses of patients in the reproductive phase with a history of recent pregnancy having acute abdomen with hemoperitoneum.
The authors have stated explicitly that there are no with this any financial support or relationships that may pose potential conflict of interest in this article.
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