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Introduction Choriocarcinoma syndrome with multiple lung metastases has a poor prognosis and causes respiratory failure due to alveolar hemorrhage. We encountered a case where the introduction of extracorporeal membrane oxygenation effectively sustained oxygenation until chemotherapy took effect on lung metastases of testicular tumors. Case presentation A 35‐year‐old man with dyspnea was referred to our hospital. He showed left testicular tumor with multiple lung metastases. Serum human chorionic gonadotropin level was also elevated. Reduced chemotherapy was initiated and extracorporeal membrane oxygenation was administered because of low oxygen levels on the fourth day. Chemotherapy successfully reduced the size of the lung masses, and extracorporeal membrane oxygenation was discontinued. Respiratory status improved substantially, but the patient died of brain metastases 4 months later. Conclusion Extracorporeal membrane oxygenation may be a useful option for managing respiratory failure resulting from choriocarcinoma syndrome until the respiratory condition is improved by chemotherapy for testicular tumors.

Gestational choriocarcinoma is a malignant trophoblastic tumor arising from any gestational event, even with a long latency period, generally in the reproductive female. It is associated with a high level of beta-human chorionic gonadotropin. Its primary site is usually the uterus but not all patients have a detectable lesion in this site. Regression of the primary tumor after it has metastasized is not uncommon, and one-third of cases manifest as complications of metastatic disease. In this report we present an uncommon case of gestational choriocarcinoma with lung, liver and jejunal metastases at the time of diagnosis without evidence of pelvic disease, in 34-year-old woman. The main points of interest of our case were the development of the ovarian hyperstimulation syndrome with massive multicystic ovarian enlargement induced by high level of beta-human chorionic gonadotropin and the bleeding of jejunal and liver metastases, due to the high vascularity of the tumor tissue, a condition known as “Choriocarcinoma Syndrome”. We will focus on the radiological findings of metastases, bleeding complications and ovarian hyperstimulation syndrome.

Choriocarcinoma (CC) is a very rare and aggressive neoplasm. The characteristic feature of this disease is a rapid hematogenous spread, mainly to the lungs and brain, which largely defines clinical signs of the disease and complicates the diagnosis. Gastrointestinal metastases are rare, and of those, only few cases with gastric location have been reported. There are publications describing choriocarcinoma syndrome (CCS). As a rule, it presents in patients with an advanced disease and is characterized by hemorrhage from metastatic foci, leading to hemoptysis and gastrointestinal bleeding. CCS development is associated with poor prognosis and high mortality. This article describes a case of testicular CC with rare few gastric metastases, complicated by CCS.

Background: Germ cell tumors (GCTs) represent a highly curable cancer. However, a small proportion of poor-risk patients can develop choriocarcinoma syndrome (CS) connected with acute respiratory distress syndrome (ARDS) with a high mortality rate. Our retrospective study aimed to determine the risk factors of poor-risk GCTs susceptible to CS development. Patients and Methods: Using a computerized database and a systematic chart review, we identified the records of 532 patients with GCTs treated at the National Cancer Institute from 2000 to 2018. Ninety eligible patients with poor-risk GCTs based on IGCCCG classification were identified. All patients were treated with platinum-based induction chemotherapy. Clinicopathological variables were collected and analyzed in correlation with CS development. Results: Nine (10%) of 90 patients developed CS in a median of 1 day (1–9 days) after chemotherapy administration. All patients died shortly after the chemotherapy start with a median of 4 days (3–35 days) due to ARDS development. In univariate analysis, metastatic lung involvement ≥50% of lung parenchyma, choriocarcinoma elements in histology specimen, dyspnea, cough, hemoptysis, ECOG PS ≥2, weight loss, hemoglobin ≤100 g/l, and NLR ≥3.3 at the time of presentation were associated with CS development. In multivariate analysis, ECOG PS ≥2 and metastatic lung involvement ≥50% were independently associated with CS. All patients with these two characteristics developed CS, compared to 0% with zero or one of these factors (p < 0.000001). Conclusions: In our study, we identified factors associated with CS development. These factors might improve the risk stratification of the patients susceptible to CS and improve their outcome. [ABSTRACT FROM AUTHOR]

BackgroundGerm cell tumors (GCTs) represent a highly curable cancer. However, a small proportion of poor-risk patients can develop choriocarcinoma syndrome (CS) connected with acute respiratory distress syndrome (ARDS) with a high mortality rate. Our retrospective study aimed to determine the risk factors of poor-risk GCTs susceptible to CS development.Patients and MethodsUsing a computerized database and a systematic chart review, we identified the records of 532 patients with GCTs treated at the National Cancer Institute from 2000 to 2018. Ninety eligible patients with poor-risk GCTs based on IGCCCG classification were identified. All patients were treated with platinum-based induction chemotherapy. Clinicopathological variables were collected and analyzed in correlation with CS development.ResultsNine (10%) of 90 patients developed CS in a median of 1 day (1–9 days) after chemotherapy administration. All patients died shortly after the chemotherapy start with a median of 4 days (3–35 days) due to ARDS development. In univariate analysis, metastatic lung involvement ≥50% of lung parenchyma, choriocarcinoma elements in histology specimen, dyspnea, cough, hemoptysis, ECOG PS ≥2, weight loss, hemoglobin ≤100 g/l, and NLR ≥3.3 at the time of presentation were associated with CS development. In multivariate analysis, ECOG PS ≥2 and metastatic lung involvement ≥50% were independently associated with CS. All patients with these two characteristics developed CS, compared to 0% with zero or one of these factors (p < 0.000001).ConclusionsIn our study, we identified factors associated with CS development. These factors might improve the risk stratification of the patients susceptible to CS and improve their outcome.

Background: Choriocarcinoma syndrome is rare and challenging in practice. The condition is fatal if it is associated with uncontrolled bleeding. Objectives: The current study aimed to present our experience managing a case of choriocarcinoma syndrome, which complicated a gestational trophoblastic disease. Patients and Methods: A case of 35-year-old female with a history of molar pregnancy that was missed and diagnosed with choriocarcinoma stage 4. The patient suffered from a life-threatening gastrointestinal (GIT) bleeding. Results: The patient was managed by intensive transfusion protocol and chemotherapy [EMA-EP protocol (etoposide, methotrexate, actinomycin D, etoposide, cisplatin) and intercaecal methotrexate]. Severe life threating bleeding stopped after 72 hours of active management and blood transfusion. Following six cycles of this protocol, BHCG level returned to normal level with marked improvement of metastatic lesions. Conclusion: Choriocarcinoma syndrome is a life-threatening condition that requires intensive management. Cure-rate index is high if the condition is appropriately managed. [ABSTRACT FROM AUTHOR]

Choriocarcinoma (CC) is a very rare and aggressive neoplasm. The characteristic feature of this disease is a rapid hematogenous spread, mainly to the lungs and brain, which largely defines clinical signs of the disease and complicates the diagnosis. Gastrointestinal metastases are rare, and of those, only few cases with gastric location have been reported. There are publications describing choriocarcinoma syndrome (CCS). As a rule, it presents in patients with an advanced disease and is characterized by hemorrhage from metastatic foci, leading to hemoptysis and gastrointestinal bleeding. CCS development is associated with poor prognosis and high mortality. This article describes a case of testicular CC with rare few gastric metastases, complicated by CCS. [ABSTRACT FROM AUTHOR]

Rare pediatric tumors are heterogeneous group containing a variety of histopathological diseases, they represent approximately 10% of all childhood cancers. These rare tumors had a diversity of histology and clinical behaviors that pose different challenges to the investigators. Exploring different pediatric rare tumors. The data were reviewed, retrospectively, through the medical records of seven rare pediatric diseases between 2012 and 2019. Giant cell fibroblastoma (GCF) presented as painless swelling in the trunk, positive for CD34 with PTEN gene mutation. Neuroglial heterotopic tissue presented in 7 days old girl with facial asymmetry and bulging in the oral cavity, maximal de-bulking was done, histopathology was positive for GFAP and S100p. Left side neck mass, surgically excised revealed non-metastatic salivary grand mucoepidermoid carcinoma. Follow up without any chemotherapy or radiotherapy for 5 years with complete remission. Mesenchymal chondrosarcoma (MCS) presented in maxillofacial bones by persistent nasal bleeding, HEY1-NCOA2 fusion gene confirmed the diagnosis. Extra-osseous Ewing sarcoma (EES) presented as rubbery painless swelling in the scalp with fusion transcript involving EWSR1-FL11. Juvenile xanthogranuloma (JXG) presented by butter fly like skin patch in the face with foamy histiocytes in upper dermis with few Touton giant cells, extensive systemic involvement of lung and bone marrow. Metastatic ovarian choriocarcinoma with choriocarcinoma syndrome received induction two different lines of chemotherapy and consolidated with autologous stem cell transplant. Seven pediatric rare tumors, with different aspects of challenges in diagnosis and management, despite the absence of formal protocols and rarity of other center experiences. [ABSTRACT FROM AUTHOR]

Introduction: Choriocarcinoma syndrome with multiple lung metastases has a poor prognosis and causes respiratory failure due to alveolar hemorrhage. We encountered a case where the introduction of extracorporeal membrane oxygenation effectively sustained oxygenation until chemotherapy took effect on lung metastases of testicular tumors., Case Presentation: A 35-year-old man with dyspnea was referred to our hospital. He showed left testicular tumor with multiple lung metastases. Serum human chorionic gonadotropin level was also elevated. Reduced chemotherapy was initiated and extracorporeal membrane oxygenation was administered because of low oxygen levels on the fourth day. Chemotherapy successfully reduced the size of the lung masses, and extracorporeal membrane oxygenation was discontinued. Respiratory status improved substantially, but the patient died of brain metastases 4 months later., Conclusion: Extracorporeal membrane oxygenation may be a useful option for managing respiratory failure resulting from choriocarcinoma syndrome until the respiratory condition is improved by chemotherapy for testicular tumors., Competing Interests: The authors declare no conflict of interest., (© 2024 The Authors. IJU Case Reports published by John Wiley & Sons Australia, Ltd on behalf of Japanese Urological Association.)

Introduction Choriocarcinoma syndrome is caused by bleeding from metastatic germ cell tumors with choriocarcinoma components. Here, we report a case of acute respiratory distress syndrome, which arose after first‐line chemotherapy for an extragonadal germ cell tumor without lung metastasis. Case presentation A 41‐year‐old male visited our institution with chief complaints of back pain and weight loss. Computed tomography showed multiple lymph node metastases in the retroperitoneal cavity. There were no lung metastases. A lymph node biopsy resulted in a diagnosis of choriocarcinoma. Bleomycin etoposide cisplatin therapy was started as induction chemotherapy. On the first day, he was diagnosed with acute respiratory distress syndrome due to choriocarcinoma syndrome. We administered high‐dose hydrocortisone therapy for 3 days. The patient's respiratory status improved. Conclusion In patients who are at high risk of developing choriocarcinoma syndrome, induction chemotherapy might lead to the development of acute respiratory distress syndrome due to the release of cytokines despite the absence of lung metastasis.

Testicular germ cell tumors (TGCTs) represent the most common solid tumor in young men and is a model of curable cancer. The effectiveness of cisplatin-based chemotherapy secures more than 95% of patients' 5-years survival rate. However, some high-risk patients with a very advanced disease develop choriocarcinoma syndrome (CS) connected with acute respiratory failure with poor prognosis and high mortality rate shortly after beginning systemic chemotherapy. CS was first described as a syndrome with hemorrhage from metastatic sites in patients with TGCTs with significantly high choriogonadotropin level. Acute hemorrhage to lung metastases is typical, but hemorrhage can occur from any metastatic site. Patognomic of choriocarcinoma cells is an invasion of small blood vessels within CS. The incidence of CS in patients with TGCTs are not well-defined and can vary across the world. To date, there are a few case reports and small retrospective series reporting a connection between systemic chemotherapy and the development of CS in metastatic TGCTs. CS is known to be triggered by massive tumor cell lysis as a result of chemotherapy and cytokine release, aggravated with alveolar hemorrhage. This can lead to a consecutive superinfection, furthered with neutropenia after chemotherapy, acute respiratory distress syndrome, rising to systemic inflammatory response, resulting in multiorgan failure and death. A reasonably effective approach in patients with extensive disease could be a shortened course of chemotherapy as well as a reduction of dosage in induction chemotherapy before full-dose chemotherapeutical regimen; however, current data regarding optimal treatment approach are limited. Patients' referral to tertiary centers and the administration of induction chemotherapy in an intensive care unit setting could further improve the treatment outcome.

Key Clinical Message: Acute spinal cord compression in the immediate postoperative period from a possible choriocarcinoma syndrome (CCS) on hemorrhagic epidural spinal metastasis has never been described before and needs to be promptly recognized and managed. A low hemoglobin associated with choriocarcinoma should raise suspicion of this syndrome. [ABSTRACT FROM AUTHOR]

A germ cell tumor is the most common form of malignancy in early male life, and can be classified as either seminomatous or nonseminomatous. Choriocarcinoma, comprised of nonseminomatous germ cells, is the most aggressive type of germ cell tumor and characteristically metastasizes to the retroperitoneal lymph nodes and less frequently to the lungs, liver, bone or brain [Shibuya et al., 2009;48: 551–554]. A 56-year-old man was admitted to another hospital complaining of abdominal distension. Symptoms included anorexia, vomiting, and diarrhea. The patient was diagnosed with an extragonadal germ cell tumor and referred to our hospital to receive chemotherapy. The day after admission, the patient’s abdominal distension gradually worsened. An emergency operation revealed venous hemorrhage from the surface of a metastatic extragonadal germ cell tumor between the ligament of Treitz and the inferior mesenteric vein in a horizontal position. Hemostatic treatment was performed with 4-0 proline thread attached to a medicated cotton sponge, rather than using a simple proline thread, and the closure area was manually compressed. Chemotherapy was initiated on postoperative day 10. A metastatic extragonadal germ cell tumor that causes massive hemorrhage and gastrointestinal hemorrhage is very rare, and represents a life-threatening emergency. If the patient’s condition carries a substantial risk of bleeding to death, it may be worthwhile to attempt abdominal operations.

Background: Choriocarcinoma syndrome is known as a lethal complication from tumoral hemorrhage, which frequently occurs at the site of tumor metastasis. Case presentation: A 59-year-old man with 60-pack-year smoking history was referred to our hospital because of hemoptysis. Chest computed tomography (CT) showed a 28 × 18 mm spiculated nodule with a cavity infiltrating the left upper lobe. A transbronchial lung biopsy was performed, and histopathological examinations revealed adenocarcinoma. No distant or regional metastasis was observed, and therefore, the patient underwent a left upper lobectomy with lymphadenectomy. Histological examinations showed that the tumor consisted of poorly differentiated adenocarcinoma cells and a choriocarcinomatous component; no multiple pulmonary metastases and mediastinal lymph node metastasis were observed. Immunohistochemical analysis showed a positive immunoreaction for human chorionic gonadotropin in the syncytiotrophoblastic cells of the choriocarcinoma. One month after the operation, the patient developed massive hemoptysis. CT showed diffuse alveolar infiltration in both the lungs. A bronchoscopic examination showed bleeding from the right upper bronchus. Aspiration cytology showed carcinoma. Despite blood transfusion and management in the intensive care unit, the patient died one and a half month after diagnosis. Conclusions: We herein report a case of a man who developed choriocarcinoma syndrome 1 month after resection for combined choriocarcinoma and adenocarcinoma of the lung. Choriocarcinoma syndrome is a rare and life-threatening complication which may occur in patients with primary pulmonary choriocarcinoma. However, we need to consider the risk of this syndrome while dealing with patients who have massive hemoptysis. [ABSTRACT FROM AUTHOR]

A germ cell tumor is the most common form of malignancy in early male life, and can be classified as either seminomatous or nonseminomatous. Choriocarcinoma, comprised of nonseminomatous germ cells, is the most aggressive type of germ cell tumor and characteristically metastasizes to the retroperitoneal lymph nodes and less frequently to the lungs, liver, bone or brain [Shibuya et al., 2009;48:551-554]. A 56-year-old man was admitted to another hospital complaining of abdominal distension. Symptoms included anorexia, vomiting, and diarrhea. The patient was diagnosed with an extragonadal germ cell tumor and referred to our hospital to receive chemotherapy. The day after admission, the patient's abdominal distension gradually worsened. An emergency operation revealed venous hemor-rhage from the surface of a metastatic extragonadal germ cell tumor between the ligament of Treitz and the inferior mesenteric vein in a horizontal position. Hemostatic treatment was performed with 4-0 proline thread attached to a medicated cotton sponge, rather than using a simple proline thread, and the closure area was manually compressed. Chemotherapy was initiated on postoperative day 10. A metastatic extragonadal germ cell tumor that causes massive hemorrhage and gastrointestinal hemorrhage is very rare, and represents a lifethreatening emergency. If the patient's condition carries a substantial risk of bleeding to death, it may be worthwhile to attempt abdominal operations. [ABSTRACT FROM AUTHOR]

Choriocarcinoma syndrome is a life-threatening lysis syndrome caused by blood vessel rupture and subsequent tumor bleeding. We describe a case of pretreatment choriocarcinoma syndrome that developed in a 27-year-old man. He underwent a high orchiectomy at a local hospital and was diagnosed with metastatic testicular tumor given the high serum human chorionic gonadotropin levels (943,601 mIU/mL). Thus, he was referred to our institution. Although he had bulky lung metastases and alveolar bleeding, we were able to administer full-dose chemotherapy with etoposide and cisplatin. On day 3 of chemotherapy, he presented with severe hypoxia and worsening of alveolar bleeding. Thus, he underwent tracheal intubation at the Intensive Care Unit. Full-dose chemotherapy was continued, and the patient was extubated upon improvement. He is currently alive and continuing treatment at another hospital. [ABSTRACT FROM AUTHOR]

Testicular germ cell tumors represent the most common malignancy among young men. While 5-year overall survival and cure for this population is greater than 95 %, choriocarcinoma is an aggressive subtype of this disease with far worse prognosis-5-year survival for choriocarcinoma is less than 80 %. In order to be able to treat these patients appropriately, a provider must recognize characteristic features of choriocarcinoma including elevated human chorionic gonadotropin in a young man with testicular mass; the astute clinician should also know the signs and symptoms of choriocarcinoma syndrome, characterized by bleeding from metastatic sites, which represents a medical emergency and is associated with high morbidity and mortality. Treatment should be directed towards a goal of tumor marker normalization, and patients with refractory disease should be considered for advanced therapies and clinical trials. Choriocarcinoma is a unique and aggressive germ cell malignancy, and these patients require early aggressive treatment to improve their chance of survival. [ABSTRACT FROM AUTHOR]

40-year-old man was referred to our hospital for treatment of metastatic testicular cancer. Computerized tomography revealed multiple lung, liver, and retroperitoneal lymph node metastases. In addition, magnetic resonance imaging revealed multiple brain metastases. Induction chemotherapy with bleomycin, etoposide, and cisplatin was started the day after a high orchiectomy. The pathological diagnosis of the surgical specimen was yolk sac carcinoma. The serum human chorionic gonadotropin (hCG) was markedly increased to 630,000 mIU/ml, which suggested the presence of a choriocarcinoma element at metastatic sites. The patient subsequently suffered respiratory failure due to pulmonary hemorrhage. Intensive supportive care prevented a fatal outcome. Physicians who treat advanced testicular tumors should be aware of the potential complication of acute pulmonary hemorrhage, called choriocarcinoma syndrome, in cases with a high hCG level, which indicates a rapidly progressive and high-volume choriocarcinoma. [ABSTRACT FROM AUTHOR]

Choriocarcinoma syndrome is an uncommon, potentially fatal complication of germ cell tumors (GCTs) in adults, but it is not well documented in children. Pediatric central nervous system (CNS) GCTs comprise a rare group of malignancies not usually associated with extra-CNS metastatic disease. Here, we report the case of a pediatric patient with a suprasellar mixed GCT and pulmonary metastases who presented with intratumoral hemorrhage and stroke. Choriocarcinoma syndrome developed soon after initiating chemotherapy. The primary tumor and pulmonary metastases were successfully treated using a multidisciplinary approach, including neurovascular intervention, chemotherapy, and craniospinal irradiation., (© 2021 Wiley Periodicals LLC.)

Nongestational ovarian choriocarcinoma (NGOC) accounts for <1% of ovarian germ cell tumors and may develop into the rare and fatal complication of choriocarcinoma syndrome. We reported a case of a 12-year-old girl with NGOC that metastasized to the lungs, retroperitoneal lymph nodes and brain. On day 2 of chemotherapy with actinomycin D and etoposide, choriocarcinoma syndrome developed due to a massive pulmonary hemorrhage, presenting as acute respiratory distress syndrome. The patient received mechanical ventilation and multimodal support and completed two cycles of an actinomycin D and etoposide regimen with intubation. After the patient's acute respiratory distress syndrome was under control, she received 9 cycles of more intensive chemotherapy regimens and achieved complete remission. An exploratory laparotomy with salpingo-oophorectomy confirmed ovarian choriocarcinoma. The patient remained disease-free at a 3-month follow-up visit. In conclusion, appropriate management consisting of multimodal support and timely, sequential and intensive chemotherapy is effective for NGOC complicated with choriocarcinoma syndrome. Stating with mild regimens would probably reduce the risk of choriocarcinoma syndrome, or at least lessen its severity. To our knowledge, we presented the first report of NGOC-related choriocarcinoma syndrome. [ABSTRACT FROM AUTHOR]

Testicular germ cell tumors (TGCTs) represent the most common solid tumor in young men and is a model of curable cancer. The effectiveness of cisplatin-based chemotherapy secures more than 95% of patients' 5-years survival rate. However, some high-risk patients with a very advanced disease develop choriocarcinoma syndrome (CS) connected with acute respiratory failure with poor prognosis and high mortality rate shortly after beginning systemic chemotherapy. CS was first described as a syndrome with hemorrhage from metastatic sites in patients with TGCTs with significantly high choriogonadotropin level. Acute hemorrhage to lung metastases is typical, but hemorrhage can occur from any metastatic site. Patognomic of choriocarcinoma cells is an invasion of small blood vessels within CS. The incidence of CS in patients with TGCTs are not well-defined and can vary across the world. To date, there are a few case reports and small retrospective series reporting a connection between systemic chemotherapy and the development of CS in metastatic TGCTs. CS is known to be triggered by massive tumor cell lysis as a result of chemotherapy and cytokine release, aggravated with alveolar hemorrhage. This can lead to a consecutive superinfection, furthered with neutropenia after chemotherapy, acute respiratory distress syndrome, rising to systemic inflammatory response, resulting in multiorgan failure and death. A reasonably effective approach in patients with extensive disease could be a shortened course of chemotherapy as well as a reduction of dosage in induction chemotherapy before full-dose chemotherapeutical regimen; however, current data regarding optimal treatment approach are limited. Patients' referral to tertiary centers and the administration of induction chemotherapy in an intensive care unit setting could further improve the treatment outcome.

We report a case of a man with testicular cancer metastatic to the lung and retroperitoneal lymph node, with significant elevation of serum levels of human chorionic gonadotropin, 534 000 mIU/mL. Just after the initiation of chemotherapy, life-threatening hemothorax occurred and hemorrhagic shock ensued. The pulmonary tumor had broken off, and lower lobectomy was carried out. Pathologic examination of the specimen revealed choriocarcinoma and yolk sac tumor. Through multimodal treatments he achieved complete remission. To our knowledge, this is the first case report of choriocarcinoma syndrome with life-threatening hemorrhage caused by rupture of pulmonary metastases, resulting in complete remission through multimodal treatments. [ABSTRACT FROM AUTHOR]