Postpartum choriocarcinoma - a rare cause of delayed postpartum hemorrhage: Four case reports and literature review.

BACKGROUND: Delayed postpartum hemorrhage is rare, with an incidence of 0.5% to 2.0% in all pregnancies. The most important causes are placental remnants, infections, and placental bed subinvolution. Postpartum choriocarcinoma, a highly malignant complication of pregnancy, is a rare condition that can be easily misdiagnosed as other common causes, such as gestational remnants, and delays the diagnosis. METHODS: Four patients visited our clinic complaining of delayed postpartum hemorrhage, combined with respiratory and neurological symptoms in 2 cases. Two cases were confirmed by histopathological examination and in addition, medical history, elevated human chorionic gonadotropin (hCG) level, and imaging findings help confirm the diagnosis of delayed postpartum hemorrhage caused by postpartum choriocarcinoma in other cases. Individualized combination chemotherapies were prescribed. In the light of massive cerebral metastasis in case 2, intrathecal methotrexate injection combined with whole-brain radiotherapy was prescribed. RESULTS: Due to the absence of routine monitoring of ß-hCG following full-term delivery, there was widespread metastasis at the time of diagnosis. Three patients got complete remission and there is no sign of recurrence. One patient had relapse and widespread metastasis and died at home 6 months after the last chemotherapy. CONCLUSION: It is important to be aware of the possibility of choriocarcinoma in patients with delayed postpartum hemorrhage. Clinicians should improve the recognition of choriocarcinoma following full-term delivery, emphasize the monitoring of ß-hCG, comprehensively analyze the general condition of patients, and conduct standardized and individualized chemotherapy protocols.

Gestational Trophoblastic Disease with Coexisting Progressing Pregnancy: Personalised Treatment Modalities.

Purpose: Gestational trophoblastic disease (GTD) coexisting with a steadily progressing pregnancy is an extremely rare condition presented in the literature as a single case or case series of successful delivery. The purpose of this study was to describe five cases of GTD and present possible management strategies for such patients. Methods: Clinical data of five pregnancies with coexisting GTD were identified within the Almazov National Medical Research Centre from 2018 to 2021. Results: Three cases of multiple pregnancies with complete hydatidiform moles and two cases of singleton pregnancies with intraplacental choriocarcinoma and invasive hydatidiform moles were identified. Three pregnancies were prolonged and ended with preterm deliveries. Malignant transformation of the GTD accounted for 60% of the cases. The condition of newborns was based on the level of prematurity and functional immaturity, and in all cases, it was aggravated by anemia. Conclusion: GTD coexisting with progressing pregnancy is threatened by the risks of preterm delivery, miscarriage, hemorrhage, and disease progression and requires monitoring in a multidisciplinary clinic experienced in the management of patients with malignant tumors during pregnancy. In cases of prolonged pregnancy against the background of GTD, we suggest the following monitoring during pregnancy: pelvic, abdominal ultrasound/MRI (without contrast), prenatal invasive fetal karyotype testing in cases of singleton pregnancy, lung X-ray/CT with uterine shielding, weekly assessment of ß-hCG levels, and dynamic monitoring of the fetus. The following postnatal monitoring should be performed: morphological examination of the placenta, weekly assessment of ß-hCG levels up to normalization, then monthly assessment up to six months, and control of ß-hCG level of the newborn.

Maternal and fetal outcomes of intraplacental choriocarcinoma complicated by fetomaternal hemorrhage: a systematic review.

INTRODUCTION: Intraplacental choriocarcinoma is a gestational trophoblastic neoplasia located within the placenta. Due to the usual silent presentation, more than half of the cases are diagnosed incidentally. It has been demonstrated that this pathology is linked to feto-maternal hemorrhage (FMH), stillbirth, and intrauterine growth restriction. The aim of our review was to establish if there are recurrent signs that might lead to an early diagnosis and better management in cases complicated by FMH. MATERIALS AND METHODS: We performed a systematic review of the literature from 2000 up to March 2023. The adopted research strategy included the following terms: (gestational choriocarcinoma obstetrics outcome) AND (intraplacental choriocarcinoma) AND (gestational choriocarcinoma). The MEDLINE (PubMed), Google Scholar, and Scopus databases were searched. RESULTS: The research strategy identified 19 cases of FMH coexisting with intraplacental choriocarcinoma (IC), as described in 17 studies. The perinatal mortality rate was 36.8%. In eight cases, histological diagnosis of IC was made post-delivery. Metastatic lesions were found in 75% (6/8) of described cases. One case of maternal death has been described. Chemotherapy was necessary in seven cases. Sporadical prenatal ultrasound signs were described. DISCUSSION: The diagnosis of IC is usually delayed, mostly due to aspecific symptoms and signs. Histological analysis of the placenta, when not routinely performed, should be performed when warning symptoms are encountered. The maternal prognosis was good, with a mortality rate of 5.5%. A fertility-sparing approach is always possible even in the presence of metastasis. Chemotherapy seems to be useful in cases of maternal and neonatal metastasis.

Recurrent choriocarcinoma complicated with leprosy during chemotherapy: A case report and literature review.

RATIONALE: The global prevalence of leprosy has decreased substantially, and cases of leprosy infection are extremely rare in China. In this report, we present a case of recurrent choriocarcinoma complicated by leprosy infection during chemotherapy. PATIENT CONCERNS: A 24-year-old Chinese woman (gravida 3, para 2) presented to a local hospital with vaginal bleeding. Her medical history included a previous diagnosis of hydatidiform mole. DIAGNOSES, INTERVENTIONS AND OUTCOMES: The patient was diagnosed with choriocarcinoma and received chemotherapy in 6 cycles. Shortly after the initial treatment was completed, the disease recurred twice with resistance to multiple chemotherapeutic agents. In her second recurrence of choriocarcinoma, she was diagnosed with leprosy with many cutaneous nodules throughout her entire body. The patient was administered chemical treatment for leprosy with the multidrug therapy regimen after being diagnosed. To prevent exacerbating the infection, no immunotherapy was utilized to treat cancer, and the infection was well-controlled at the conclusion of anticancer therapy. LESSONS: Because of immunological reduction, cancer patients are susceptible to a variety of infections. For patients with cancer, prevention and early detection of rare infectious diseases should receive special attention. Immunotherapy must be used with caution when treating patients with cancer and infections.

Pure testicular choriocarcinoma with gastrointestinal metastasis and paraneoplastic symptoms: a case report.

BACKGROUND: Pure testicular choriocarcinoma is a rare type of non-seminomatous germ cell tumor extremely poor prognostic with the tendency to bleed at the metastatic site. At the time of the diagnosis, 70% of patients have metastatic lesions. Depending on the site of the metastasis, symptoms vary. Gastrointestinal involvement is seen in less than 5% of cases, mostly in the duodenum. CASE PRESENTATION: We present a 47 years old male with testicular choriocarcinoma involving the jejunum, lung, liver, and kidney presenting with acute abdominal pain, melena, and dyspnea with some paraneoplastic symptoms. The patient had increased, severe and constant pain in the right lower quadrant for the previous four days. Additionally, he was complaining of nausea, vomiting, anorexia, and a history of melena for the last 10 days. Dyspnea on exertion, hemoptysis, and dry cough were the symptoms he was suffering from, for almost one year. The patient's general appearance was pale, ill, and thin with 10 kg of weight loss during the last some months. The computed tomography (CT) scan reported multiple metastatic lesions in both liver lobes and the left kidney. Pathologic study of the samples of small bowel lesions showed metastatic choriocarcinoma. Following the patient had been referred to an oncologist to start the chemotherapy regime. Finally, the patient has expired after 40 days of his first admission. CONCLUSIONS: Testicular choriocarcinoma is a rare but fatal malignancy among young men. Gastrointestinal metastases are infrequent involvement represented by melena and acute abdominal pain, obstruction, and mass. Physicians should consider it as a differential diagnosis for acute abdomen and gastrointestinal bleeding causation.

A rare case of gestational ovarian choriocarcinoma coexistent with intrauterine pregnancy.

OBJECTIVE: To report the rare case of gestational primary ovarian choriocarcinoma coexistent with intrauterine pregnancy, successfully treated with surgery and systemic chemotherapy. We also describe the utility of short tandem repeat (STR) genotyping in the diagnosis of choriocarcinoma. CASE REPORT: A 38-year-old woman at 17 gestational weeks presented with an ovarian tumor rupture in the left ovary. Left salpingo-oophorectomy was performed and the patient was diagnosed with gestational ovarian choriocarcinoma via histopathology and STR genotyping. After artificial abortion, the patient underwent 8 cycles of chemotherapy. Abdominal hysterectomy was performed because of the presence of low levels of human chorionic gonadotropin and the tumor that developed behind the uterus. However, no viable choriocarcinoma cells were found in the residual tumor, suggesting that the patient achieved full remission. CONCLUSIONS: Early detection is crucial in treating choriocarcinomas; thus, clinicians should consider the possibility of choriocarcinoma at the presence of an ovarian tumor during pregnancy. Gestational and non-gestational choriocarcinomas differ in prognosis and sensitivity to chemotherapy due to their different etiologies. Therefore, STR genotyping may be beneficial in predicting the patient's prognosis or selecting the appropriate regimen.

Gastrointestinal bleeding caused by metastatic testicular choriocarcinoma: a case report and literature review.

BACKGROUND: Testicular tumor is one of the common solid tumors in young men. Testicular choriocarcinoma is a non-spermatogonial germ cell tumor, which is the rarest of all testicular cancers. Choriocarcinoma usually shows bleeding at the metastatic site, while gastrointestinal involvement is rare. METHODS: Here, we report a case of testicular choriocarcinoma with gastrointestinal bleeding as the first diagnosis and summarize the similar cases all over the world in recent 20 years. RESULTS: A 28-year-old male was treated with repeated melena for 2 months. No bleeding foci of the stomach, duodenum, colon, and rectum were found in endoscopy, and no bleeding foci of digestive tract was found in selective angiography, but a space occupying lesions of the lung, liver, and upper jejunum were found in chest and abdominal CT. Considering the possibility of a metastatic tumor and the ineffectiveness of medical treatment, the patient was converted to surgical treatment. The postoperative pathology was consistent with testicular choriocarcinoma. The patient received a chemotherapy regimen of paclitaxel, ifosfamide, and cisplatin. At present, the chemotherapy regimen is well tolerated. CONCLUSIONS: The case report confirmed that even if we cannot find the logical relationship between clinical manifestations and genital examination, genital examination should also be part of the patient's systematic examination.

Severe thyrotoxicosis as initial presentation of gastric choriocarcinoma: a case report.

BACKGROUND: Extragonadal choriocarcinoma is rare and can be associated with hyperthyroidism when producing very high levels of human chorionic gonadotropin. CASE PRESENTATION: A 62-year-old Hispanic female presented with a 3-week history of shortness of breath, palpitations, extreme weakness, new-onset hot flashes, and right flank pain. Her physical examination was remarkable for tachycardia, hepatomegaly, hyperreflexia, and tremor; goiter was absent. Laboratory studies revealed increased lactate dehydrogenase, alkaline phosphatase, suppressed thyroid stimulating hormone, very elevated T4, and absent thyroid stimulating immunoglobulin. 18F-fluorodeoxyglucose positron emission tomography-computed tomography exhibited hepatomegaly with multiple large fluorodeoxyglucose-avid liver masses and a focus of fluorodeoxyglucose avidity in the stomach with no structural correlate. A thyroid scan (99mTcO 4 - ) showed diffusely increased tracer uptake. She was started on propranolol and methimazole. Upon stabilization of severe thyrotoxicosis, upper endoscopy was performed, showing a ~ 5 cm bleeding lesion in the greater stomach curvature body; biopsy was consistent with choriocarcinoma; beta-human chorionic gonadotropin hormone was 2,408,171 mIU/mL. The patient received methotrexate followed by etoposide and cisplatin. Methimazole was titrated down, and upon liver failure the medication was stopped. The thyrotoxicosis was effectively controlled with antithyroid drug and concurrent chemotherapy. At ~ 1.5 months after initial diagnosis, the patient died due to bleeding/acute liver failure with coagulation defects followed by multiple organ failure. CONCLUSIONS: Severe thyrotoxicosis can represent an unusual initial presentation of metastatic choriocarcinoma in the setting of extreme elevation of beta-human chorionic gonadotropin. Primary gastric choriocarcinoma is an aggressive malignancy with very poor outcomes. The co-occurrence of severe thyrotoxicosis with advanced primary gastric choriocarcinoma and imminent liver failure complicates management options.

Pretreatment of choriocarcinoma syndrome: a rare complication of metastatic germ cell tumours.

A 27-year-old man newly diagnosed with metastatic testicular choriocarcinoma developed a large right perinephric haematoma secondary to a metastatic deposit in his right kidney. His presentation was also complicated by bilateral iliac venous thrombosis and pulmonary embolism identified prior to initiation of chemotherapy. He underwent multiple attempts at angioembolisation of the bleeding vessels and ultimately angioembolisation of the main renal artery had to be performed to control the bleeding. Following resolution and commencement of chemotherapy, the patient also developed spontaneous intracranial haemorrhage requiring craniotomy.

Non-gestational uterine choriocarcinoma inside a leiomyoma: importance of early suspicion in prognosis.

Uterine choriocarcinoma is a trophoblastic neoplasm most commonly related to pregnancy. However, there are already cases of non-gestational uterine choriocarcinoma (NGC) described in literature. The difficult diagnosis may delay treatment and threaten patient survival. We report the case of a 37-year-old patient presenting with abnormal haemorrhage and moderate levels of beta-human chorionic gonadotropin (ß-hCG). As imaging exams did not show any suspicious site for the origin, choriocarcinoma was not considered so the treatment was delayed. Anatomopathological diagnosis was given both from the surgical sample of hysterectomy and from a skin lesion. The lesion was discovered inside a myoma. At this point, ß-hCG levels were substantially high and she presented with respiratory distress due to pulmonary metastasis. The patient was transferred to an intensive care unit and underwent mechanical ventilation as well as life-saving chemotherapy. At 10 months after diagnosis, she is well and in remission. The delay from not suspecting the diagnosis was almost fatal for the patient.

Fetomaternal hemorrhage: a clue to intraplacental choriocarcinoma and neonatal malignancy.

Fetomaternal hemorrhage (FMH) is a known cause of neonatal anemia due to fetal blood loss to the maternal circulation, occurring when the maternal-fetal barrier is disrupted. Several causes must be considered, although in most cases the etiology remains unknown. Intraplacental choriocarcinoma (ICC) is a rare entity and has been related with massive FMH, intrauterine fetal demise, severe neonatal anemia and metastatic choriocarcinoma in both mother and infant. There are 25 cases of histopathologically confirmed ICC complicated with FMH described in the literature. Because FMH occurs unexpectedly and the majority of patients with ICC are asymptomatic, this diagnosis may be missed. Once FMH is confirmed, underlying malignancy should be kept in mind. The authors present a case report of severe neonatal anemia following FMH related to ICC and highlight the importance of serum ß-hCG monitoring in cases of massive FMH.

Choriocarcinoma in a viable pregnancy with the rare presentation of intractable lower back pain.

OBJECTIVE: We present a case of choriocarcinoma in a viable pregnancy with the rare presentation of intractable lower back pain. CASE REPORT: The patient is a 34-year-old multiparous woman with her second pregnancy, and a history of scoliosis with spinal fixation. Her first pregnancy was uneventful, with a term vaginal delivery. She was hospitalized four times due to intractable back pain from 25 to 31 weeks, and terminated at 31 weeks. The placenta was unremarkable on gross examination. Postpartum, the patient developed obstructive ileus, requiring a rectosigmoid resection. She was diagnosed with metastatic choriocarcinoma to the liver, para-aortic lymph nodes, and mesentery. A week later, she developed micro-thrombosis of all limbs, massive ascites, pleural effusion. Patient refused chemotherapy and died on post-operative Day 15. CONCLUSION: Presentation of choriocarcinoma in pregnancy varies widely. Clinicians should consider the differential diagnosis of choriocarcinoma when faced with abnormal unexplained symptoms during pregnancy.

Three atypical presentations of choriocarcinoma, occurring during and shortly after a coexistent viable pregnancy.

Gestational choriocarcinoma is a malignant tumour originating from the trophoblastic tissue that can arise during or after any type of pregnancy, but most of the time follows a molar pregnancy. Characteristic for this tumour is its rapid haematogenous spread to various organs, causing atypical presentations often attributable to metastatic disease. We review three cases that occurred during and shortly after a coexistent intrauterine pregnancy. The patient of Case 1 presented with neurological symptoms due to hypercalcaemia, in Case 2 there was initially suspicion of appendicitis and the third patient presented with acute respiratory insufficiency. This case series illustrates that, although highly effective chemotherapy is available, choriocarcinoma can be life-threatening and accurate diagnosis is challenging but critical.

Complete remission of choriocarcinoma with pulmonary vein thrombosis in the third trimester of pregnancy treated with systemic chemotherapy and anticoagulation: A case report.

RATIONALE: Choriocarcinoma is a highly aggressive tumor. It occurs infrequently during pregnancy. The management of choriocarcinoma during pregnancy poses several challenges. PATIENT CONCERNS: At 34 weeks of gestation, a 21-year-old primigravida was transferred to the emergency room for cephalgia, reduced fetal movements, and left intra-atrial intracavitary thrombus. DIAGNOSIS: Choriocarcinoma in the third trimester with lung and brain metastases, pulmonary vein thrombosis (PVT), and systemic thrombosis. INTERVENTION: An emergency cesarean section was performed. Subsequently, low-molecular-weight heparin anticoagulation combined with multiagent chemotherapy was administered. OUTCOME: A 1.59 kg live female was born. Multiagent chemotherapy combined with anticoagulation led to complete regression of the cerebral and pulmonary lesions and the dissolution of pulmonary vein thrombus. At the 11-month follow-up, the patient remained in complete remission without complications, and her child was disease-free. LESSONS: This is the first case of gestational choriocarcinoma with PVT. Our case suggests that conservative therapy can be the first choice for small, asymptomatic PVT secondary to choriocarcinoma.

Loss of Reproductive Ability due to Late Application of Young Refugee with Choriocarcinoma During the COVID-19 Pandemic.

Choriocarcinoma is an aggressive malignant trophoblastic neoplasm. The rapid growth of neoplastic tissue and myometrial invasion can cause uterine perforation. It is important to quickly diagnose the disease and plan treatment because these tumors are nearly always curable, and fertility can be preserved in most cases with proper management. The outbreak of novel coronavirus disease has affected the whole world since January 2019 and caused delays in treatment and follow-up of patients all over the world. In this study, we report a case of choriocarcinoma who postponed her admission to the hospital after diagnosis because she was afraid of the pandemic and lost her fertility due to uterine rupture and massive intra-abdominal hemorrhage.

Non-myxoma neoplastic cerebral aneurysms: A systematic review.

Neoplastic cerebral aneurysms (NCAs) are highly rare lesions characterized by invasion of cancerous cells within the wall of an artery leading to aneurysm formation. While NCAs caused by myxomas are well characterized in the clinical literature, rarer etiologies have also been reported and are typically associated worse clinical outcomes. We performed the first PRISMA-compliant systematic literature review of true, non-myxoma neoplastic cerebral aneurysms using the PubMed/MEDLINE, Embase, Scopus, and Google Scholar databases. Data of interest included age, sex, aneurysm size, number of aneurysms, aneurysm location, neoplasm type, aneurysm treatments, cancer treatments, risk of rupture, intracerebral hemorrhage prevalence, subarachnoid hemorrhage prevalence, and survival at 90 and 180 days. A total of 50 studies met our inclusion criteria. The mean age of the patient population was 37.4 years (SD: ±16.8) and had an overall female preponderance (39/50, 78%). Of these NCA cases, 29/50 (58.0%) were choriocarcinomas, 10/50 (20.0%) were related to lung cancer, and 11/50 (22.0%) had other origins of variable pathologies. 90-day survival rates were 60.0% (15/25) for choriocarcinomas, 28.6% (2/7) for the lung cancer group, and 14.3% (1/7) for the other origins group. 180-day survival rates were 52.0% (13/25) for the choriocarcinoma group, 14.3% (1/7) for the lung cancer group, and 0% (0/7) for the other origins group. Prognosis of NCA patients ultimately depends on the course of disease progression and cancer management. Further research is needed to better understand optimal treatment modalities for patients with NCAs.

Double trouble: Extrauterine epithelioid trophoblastic tumor with uterine choriocarcinoma - An autopsy report.

Gestational trophoblastic tumors (GTTs) include choriocarcinoma, epithelioid trophoblastic tumor, and placental site trophoblastic tumor. The occurrence of mixed GTT is rare. We report such a case in a 24-year-old woman who presented with menorrhagia since 2 months and obstetric history of two abortions, one of which was a molar pregnancy. She was undergoing evaluation for carcinoma cervix and treatment for pulmonary tuberculosis from another hospital when she was admitted at our institute for further workup and treatment. However, she succumbed and an autopsy was performed. Histologic evaluation after the autopsy revealed uterine choriocarcinoma with metastatic epithelioid trophoblastic tumor (ETT) in the lung and spleen.

Epithelioid trophoblastic tumor coexisting with choriocarcinoma around an abdominal wall cesarean scar: a case report and review of the literature.

BACKGROUND: Mixed gestational trophoblastic neoplasms are extremely rare and comprise a group of fetal trophoblastic tumors including choriocarcinomas, epithelioid trophoblastic tumors, and placental site trophoblastic tumors. We present a case of a patient with extrauterine mixed gestational trophoblastic neoplasm adjacent to the abdominal wall cesarean scar. On the basis of a literature review, this type of case has never been reported before due to the unique lesion location and low incidence. CASE PRESENTATION: Our patient was a 39-year-old Chinese woman who had a history of two cesarean sections and one miscarriage. She had a recurrent anterior abdominal wall mass around her cesarean scar, and the mass was initially suspected of being choriocarcinoma of unknown origin. The patient had concomitant negative or mildly increased serum ß-human chorionic gonadotropin at follow-up and no abnormal vaginal bleeding or abdominal pain. However, she underwent local excision twice and had two courses of chemotherapy with an etoposide and cisplatin regimen. She finally opted for exploratory laparotomy with abdominal wall lesion removal, subtotal hysterectomy, bilateral salpingectomy, and left ovarian cyst resection, which showed the abdominal wall lesion, whose components were revealed by microscopy and immunohistochemical staining to be approximately 90% epithelioid trophoblastic tumors and 10% choriocarcinomas from a solely extrauterine mixed gestational trophoblastic neoplasm around an abdominal wall cesarean scar. CONCLUSIONS: It is worth noting whether epithelioid trophoblastic tumor exists in the setting of persistent positive low-level ß-human chorionic gonadotropin. More studies are required to provide mechanistic insights into these mixed gestational trophoblastic neoplasms.