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1.
J Med Case Rep ; 18(1): 323, 2024 Jul 15.
Article in English | MEDLINE | ID: mdl-39004746

ABSTRACT

BACKGROUND: Diagnosing non-gestational uterine choriocarcinoma in children is challenging because of its rarity and nonspecific imaging findings. Herein, we report a case of non-gestational uterine choriocarcinoma in a child, which was unexpectedly found during exploratory laparotomy and confirmed by histopathological findings. However, the tumor did not respond to chemotherapy. CASE PRESENTATION: A 4-year-old Indonesian female patient was brought into the emergency unit with chief complaint of vaginal bleeding. She had suffered from vaginal spotting 4 months before being admitted to the hospital. Physical examination revealed a distended abdomen in the left lumbar region and a palpable fixed mass with a smooth surface. Abdominal computed tomography scans revealed a large mass (10 × 6 × 12 cm) with fluid density and calcification. Thus, we suspected left ovarian teratoma. The patient's luteinizing hormone, follicle-stimulating hormone, and lactate dehydrogenase levels were 25.2 mIU/ml, 0.1 mIU/ml, and 406 U/l, respectively. According to the clinical and radiological findings, we decided to perform an exploratory laparotomy and found a tumor originating from the uterus, not the ovarium. We did not observe liver nodules and any enlargement of abdominal lymph nodes. Subsequently, we performed hysterectomy. The histopathological findings supported the diagnosis of choriocarcinoma. The patient was discharged uneventfully on postoperative day 5. Thereafter, the patient underwent nine cycles of chemotherapy, including carboplatin (600 mg/m2 IV), etoposide (120 mg/m2 IV), and bleomycin (15 mg/m2 IV). However, on the basis of the clinical findings of a palpable mass and partial intestinal obstruction, the tumor relapsed soon after the ninth cycle of chemotherapy. Currently, the patient is undergoing chemotherapy again. CONCLUSIONS: Although pure non-gestational uterine choriocarcinoma is rare, it should be considered as one of the differential diagnoses for intraabdominal tumors in a child, so as to better guide and counsel families regarding the surgical plan and prognosis, respectively. In the present case, the patient's response to chemotherapy was poor, implying that the treatment of non-gestational choriocarcinoma is still challenging, particularly in the pediatric population.


Subject(s)
Choriocarcinoma, Non-gestational , Hysterectomy , Uterine Neoplasms , Humans , Female , Uterine Neoplasms/diagnosis , Uterine Neoplasms/surgery , Uterine Neoplasms/pathology , Uterine Neoplasms/drug therapy , Uterine Neoplasms/therapy , Child, Preschool , Choriocarcinoma, Non-gestational/diagnosis , Choriocarcinoma, Non-gestational/pathology , Choriocarcinoma, Non-gestational/drug therapy , Choriocarcinoma, Non-gestational/therapy , Tomography, X-Ray Computed , Diagnosis, Differential , Laparotomy , Uterine Hemorrhage/etiology , Etoposide/therapeutic use , Etoposide/administration & dosage
2.
Int J Gynecol Cancer ; 34(6): 926-934, 2024 Jun 03.
Article in English | MEDLINE | ID: mdl-38123189

ABSTRACT

Choriocarcinoma is a highly vascular and invasive tumor of anaplastic trophoblast, predominantly made up of cytotrophoblasts and syncytiotrophoblasts without villi. Based on its origin, choriocarcinoma can be either gestational or non-gestational. Non-gestational choriocarcinoma can be of germ cell origin, or can be seen in association with a somatic high-grade malignancy. It is difficult to differentiate gestational from non-gestational choriocarcinoma, especially in the reproductive age group. It is important to distinguish between the two, for accurate staging and prognostication, deciding the primary treatment modality, (ie, surgery or chemotherapy), and tailoring follow-up timeframes after diagnosis. An extensive literature search was performed regarding all cases of non-gestational choriocarcinoma, published before March 2023. A note was made of whether the origin of choriocarcinoma was ascertained and how gestational choriocarcinoma was differentiated from non-gestational choriocarcinoma. The keywords used for literature search were "non-gestational choriocarcinoma", "primary choriocarcinoma", "ovarian choriocarcinoma", "ovarian germ cell tumors", or "choriocarcinomatous differentiation". This review aims to summarize the similarities and differences in the epidemiology, pathogenesis, clinical presentation, and management guidelines between gestational and non-gestational choriocarcinoma, which can form an important educational resource for clinicians and laboratory physicians dealing with such cases.


Subject(s)
Choriocarcinoma, Non-gestational , Humans , Female , Pregnancy , Choriocarcinoma, Non-gestational/diagnosis , Choriocarcinoma, Non-gestational/pathology , Choriocarcinoma, Non-gestational/therapy , Choriocarcinoma/diagnosis , Choriocarcinoma/pathology , Choriocarcinoma/therapy , Ovarian Neoplasms/diagnosis , Ovarian Neoplasms/pathology , Ovarian Neoplasms/therapy , Uterine Neoplasms/pathology , Uterine Neoplasms/diagnosis
4.
J Pediatr Adolesc Gynecol ; 34(2): 231-233, 2021 Apr.
Article in English | MEDLINE | ID: mdl-33189896

ABSTRACT

BACKGROUND: Nongestational choriocarcinoma is a rare ovarian malignancy with a prognosis worse than that of gestational choriocarcinoma. Debulking surgery is the primary treatment for ovarian carcinoma. However, fertility preservation is important in young women. CASE: A 15-year-old girl with no sexual experience was admitted for abnormal uterine bleeding. Ultrasonography showed a solid mass in the right ovary and her serum ß-human chorionic gonadotrophin levels were markedly elevated. We performed right oophorectomy, omentectomy, and peritoneal washing cytology. The uterus and left adnexa were preserved. She was diagnosed with nongestational choriocarcinoma, stage IIA. She received adjuvant chemotherapy (etoposide, methotrexate, actinomycin, cyclophosphamide, and oncovin regimen) and has been disease-free for more than 5 years. SUMMARY AND CONCLUSION: Fertility-sparing surgery combined with chemotherapy is an acceptable treatment option for young patients with locally advanced nongestational choriocarcinoma.


Subject(s)
Antineoplastic Combined Chemotherapy Protocols/therapeutic use , Choriocarcinoma, Non-gestational/therapy , Cytoreduction Surgical Procedures/methods , Ovarian Neoplasms/therapy , Adolescent , Chemotherapy, Adjuvant , Choriocarcinoma, Non-gestational/pathology , Female , Fertility Preservation , Humans , Ovarian Neoplasms/pathology , Pregnancy , Prognosis , Treatment Outcome
5.
J Oncol Pharm Pract ; 26(4): 989-994, 2020 Jun.
Article in English | MEDLINE | ID: mdl-31547751

ABSTRACT

INTRODUCTION: Primary choriocarcinoma of the colon is an extremely rare neoplasm which has a poor prognosis. Only 18 cases have been previously reported in English medical literature. Here we present a case of primary rectal choriocarcinoma with a good response to chemotherapy and review the literature on this uncommon tumor. CASE REPORT: A 36-year-old woman presented with abdominal pain and vaginal bleeding. Abdominal magnetic resonance imaging revealed 6.9 × 5.3 × 6.4 cm hypervascular mass posterior to uterus very close to rectum. Beta-human chorionic gonadotropin (ß-hCG) level was markedly elevated. Low anterior resection of the rectum with lymph node dissection and total abdominal hysterectomy with bilateral salpingo-oophorectomy were performed. Pathologic diagnosis was reported as colonic choriocarcinoma with a focal component of adenocarcinoma. Post-operative magnetic resonance imaging detected multiple metastatic lesions throughout the liver. The patient was treated with systemic chemotherapy using bleomycin, etoposide and cisplatin (BEP protocol). After three cycles, ß-hCG level decreased to normal and magnetic resonance imaging showed regression of liver metastasis. However, the patient died of respiratory failure due to bleomycin toxicity and pneumonia accompanied by rapid disease progression. DISCUSSION: This is an extremely rare case of primary rectal choriocarcinoma. Due to poor prognosis of the disease, it seems very important to start prompt treatment to improve patient's survival.


Subject(s)
Choriocarcinoma, Non-gestational/diagnostic imaging , Choriocarcinoma, Non-gestational/therapy , Rectal Neoplasms/diagnostic imaging , Rectal Neoplasms/therapy , Adult , Antineoplastic Combined Chemotherapy Protocols/administration & dosage , Bleomycin/administration & dosage , Cisplatin/administration & dosage , Etoposide/administration & dosage , Fatal Outcome , Female , Humans , Magnetic Resonance Imaging/methods , Rectum/diagnostic imaging , Rectum/surgery
6.
Cancer Biol Ther ; 20(9): 1176-1182, 2019.
Article in English | MEDLINE | ID: mdl-31132027

ABSTRACT

Nongestational choriocarcinoma is rare, especially in postmenopausal women. It may be derived from the transformation of germ cells or dedifferentiation of other tumor tissue cells. It is usually found in the ovaries but rarely in the uterus. Pure nongestational uterine choriocarcinoma in postmenopausal women is exceptional. Thirty-four cases of choriocarcinoma have been found during the past 25 y at the Tianjin Medical University General Hospital. We retrospectively reviewed the medical records of these 34 cases and found only two cases of nongestational uterine choriocarcinoma, both of which were postmenopausal women. We also reviewed 19 other cases previously reported during the past 50 y (from 1970 to 2018) that were identified as uterine choriocarcinoma in postmenopausal women. Analysis of these combined data indicates that nongestational choriocarcinoma is a rare neoplasm in postmenopausal patients. It genetically originates entirely in the patient, and short tandem repeat (STR) analyses are usually required to differentiate gestational and nongestational choriocarcinoma. Although nongestational choriocarcinoma shows some response to chemotherapy, sensitivity is much poorer than that in gestational choriocarcinoma. The prognosis for nongestational choriocarcinoma is poor and the long-term survival rate is low.


Subject(s)
Choriocarcinoma, Non-gestational/diagnosis , Choriocarcinoma, Non-gestational/therapy , Postmenopause , Biopsy , Choriocarcinoma, Non-gestational/etiology , Combined Modality Therapy , Disease Management , Disease Susceptibility , Female , Humans , Immunohistochemistry , Microsatellite Repeats , Middle Aged , Symptom Assessment , Ultrasonography
7.
Am J Dermatopathol ; 41(1): 50-54, 2019 Jan.
Article in English | MEDLINE | ID: mdl-29944473

ABSTRACT

Testicular choriocarcinoma needs to be considered in the differential diagnosis of cutaneous metastases in young adult men because of its propensity for early hematogenous dissemination. Furthermore, the diagnosis may not be suspected in many cases in which there is clinically no testicular enlargement. This highly aggressive germ cell tumor typically metastasizes to the liver, lungs, and brain. Skin metastasis is exceedingly rare with only 22 cases previously reported in the world literature. We herein report 2 additional cases: a 25-year-old man and a 32-year-old man, both of whom were treated for mixed germ cell tumors and developed multiple cutaneous metastases to the head.


Subject(s)
Brain Neoplasms/secondary , Choriocarcinoma, Non-gestational/secondary , Skin Neoplasms/secondary , Testicular Neoplasms/pathology , Adult , Biomarkers, Tumor/blood , Biopsy , Brain Neoplasms/blood , Brain Neoplasms/diagnostic imaging , Brain Neoplasms/therapy , Choriocarcinoma, Non-gestational/blood , Choriocarcinoma, Non-gestational/diagnostic imaging , Choriocarcinoma, Non-gestational/therapy , Chorionic Gonadotropin/blood , Humans , Immunohistochemistry , Magnetic Resonance Imaging , Male , Skin Neoplasms/blood , Skin Neoplasms/therapy , Testicular Neoplasms/blood , Testicular Neoplasms/therapy , Treatment Outcome
8.
Am J Surg Pathol ; 42(8): 1113-1120, 2018 08.
Article in English | MEDLINE | ID: mdl-29794869

ABSTRACT

The clinical significance of limited choriocarcinoma in a malignant mixed germ cell tumor (MGCT) is unknown. Men with a MGCT with ≤5% choriocarcinoma at radical orchiectomy (RO) between 2000 and 2016 from our consult service were studied. Of 50 men in our cohort, we had clinical information for 30 men. Median follow-up was 41 months (1 to 168 mo). Median tumor size was 4.5 cm (1.1 to 8.0 cm). In total, 22/30 (73%) cases were pT1, 6/30(20%) cases were pT2, and 2/30 (7%) cases were pT3. In total, 4/30(13%) cases had lymph node metastases and 2/30 (7%) cases had distant metastases at the time of RO. In 30 cases with RO we had information on immediate postorchiectomy treatment: 14/30 (46.7%) active surveillance, 4/30 (13.3%) retroperitoneal lymph node dissection, 10/30 (33.3%) chemotherapy (chemotherapy), 1/30 (3.3%) retroperitoneal lymph node dissection followed by chemotherapy, and 1/30 (3.3%) resection of a distant metastasis. Preoperative serum human chorionic gonadotropin (hCG) levels ranged between 0.1 and 60,715 mIU/mL (mean, 4796; median, 485). One patient had an hCG level of 6367 mIU/mL and another 60,715 mIU/mL with the remaining cases <5000 mIU/mL. In total, 4/30 (13%) patients had elevated serum markers after surgery, 3 of them normalized following chemotherapy while the fourth one continued to have elevated serum alpha fetoprotein levels after chemotherapy. All patients were alive at last follow-up. In total, 7/30 (23.3%) patients subsequently developed metastatic disease to lymph nodes or distal organs, the histology of the metastasis consisted mainly of teratoma and yolk sac tumor. Embryonal carcinoma was present in 2 metastatic sites. One lung metastasis was suggestive for choriocarcinoma. Definitive choriocarcinoma was not present in any of the metastasis. A small component of choriocarcinoma in a MGCT is typically associated with relatively low-level elevations of serum hCG levels, and is not associated with aggressive disease. The presence of limited choriocarcinoma (≤5%) does not add to the prognostic information provided by standard TNM staging, which uses levels of serum markers (hCG, alpha fetoprotein, lactate dehydrogenase) as surrogates for extent of disease.


Subject(s)
Choriocarcinoma, Non-gestational/secondary , Neoplasms, Complex and Mixed/secondary , Neoplasms, Germ Cell and Embryonal/secondary , Testicular Neoplasms/pathology , Adolescent , Adult , Chemotherapy, Adjuvant , Choriocarcinoma, Non-gestational/blood , Choriocarcinoma, Non-gestational/therapy , Chorionic Gonadotropin/blood , Humans , Lymph Node Excision , Lymphatic Metastasis , Male , Metastasectomy , Middle Aged , Neoplasm Staging , Neoplasms, Complex and Mixed/blood , Neoplasms, Complex and Mixed/therapy , Neoplasms, Germ Cell and Embryonal/blood , Neoplasms, Germ Cell and Embryonal/therapy , Orchiectomy , Testicular Neoplasms/blood , Testicular Neoplasms/therapy , Time Factors , Treatment Outcome , Tumor Burden , Young Adult , alpha-Fetoproteins/metabolism
9.
BMC Cancer ; 18(1): 543, 2018 May 08.
Article in English | MEDLINE | ID: mdl-29739453

ABSTRACT

BACKGROUND: Choriocarcinoma is a rare malignant germ-cell tumour, most commonly found in adult women. It infrequently presents as spontaneous renal haemorrhage (SRH). Genital malformation and SRH secondary to choriocarcinoma has previously been only reported in females. We present what we believe to be the first case of a male patient with genital malformation (hypospadias and cryptorchidism) and SRH at presentation of choriocarcinoma. CASE PRESENTATION: A 25-year-old man presented to the department with intense pain in the right flank region and lower back. Initial investigations showed spontaneous renal haemorrhage, for which an emergency partial nephrectomy was performed. Clinical, radiological, and pathological investigations suggested a diagnosis of testicular choriocarcinoma with metastases to the right kidney, both lungs, and brain. Initial treatment was with a chemotherapy regimen of cisplatin, etoposide and bleomycin and whole brain radiotherapy; however, 6 months after diagnosis the patient developed liver metastasis, after which time the BEP protocol was switched to ITP with oral apatinib. Despite best efforts, the liver and lung metastasis continued to grow and a decision was made to discontinue active treatment and provide only palliative care until the patient passed away. CONCLUSION: Choriocarcinoma is a difficult cancer to diagnose pre-operatively. In male patients with early metastasis, prognosis may be much poorer than in the commoner gestational choriocarcinoma. A multidisciplinary with comprehensive post-surgical intervention is of great importance in the treatment of these patients.


Subject(s)
Choriocarcinoma, Non-gestational/complications , Cryptorchidism/etiology , Hemorrhage/etiology , Hypospadias/etiology , Kidney Diseases/etiology , Testicular Neoplasms/complications , Adult , Choriocarcinoma, Non-gestational/diagnosis , Choriocarcinoma, Non-gestational/secondary , Choriocarcinoma, Non-gestational/therapy , Fatal Outcome , Hemorrhage/surgery , Humans , Kidney Diseases/surgery , Liver Neoplasms/diagnosis , Liver Neoplasms/secondary , Liver Neoplasms/therapy , Lung Neoplasms/diagnosis , Lung Neoplasms/secondary , Lung Neoplasms/therapy , Male , Nephrectomy , Palliative Care , Testicular Neoplasms/diagnosis , Testicular Neoplasms/pathology , Testicular Neoplasms/therapy
10.
Am J Surg Pathol ; 41(12): 1593-1606, 2017 Dec.
Article in English | MEDLINE | ID: mdl-28877059

ABSTRACT

Choriocarcinoma is an uncommon malignant neoplasm, which can be either gestational or nongestational in origin. Distinction of these subtypes has prognostic and therapeutic implications. Twenty-two tumors were genotyped using polymerase chain reaction amplification of 15 short tandem repeat loci and the amelogenin locus (XY determination). DNA patterns from tumor and maternal tissue, as well as villous tissue from any available prior or concurrent gestation, were compared, to determine gestational versus nongestational nature (containing vs. lacking a paternal chromosome complement, respectively) and the relationship between the tumor and any prior or concurrent gestation. Nineteen tumors were gestational. Of these, 14 were purely androgenetic/homozygous XX: 6 uterine tumors with a concurrent or prior genetically related complete hydatidiform mole (CHM), 4 uterine tumors without an accompanying villous component, 1 uterine cornual tumor separate from a genetically distinct second trimester intrauterine placenta, 1 ectopic ovarian tumor separate from a genetically distinct third trimester intrauterine placenta, and 2 ectopic fallopian tube tumors. Five gestational tumors were biparental: 3 (2 XX, 1 XY) intraplacental choriocarcinomas genetically related to the placenta and 2 uterine tumors without accompanying placental tissue after term deliveries (1 XX 4 weeks postpartum and 1 XYY with allelic imbalances 1 year postpartum; prior placentas not available for analysis). Three tumors were nongestational: all XX with allelic imbalances; 2 ovarian, 1 pelvic. Gestational choriocarcinoma can be androgenetic or biparental. Most are androgenetic/homozygous XX, often associated with a genetically related concurrent or prior CHM, and thus of molar-associated type. These findings support that homozygous XX CHMs are associated with some risk of significant gestational trophoblastic disease. Intraplacental choriocarcinomas are biparental and genetically related to the placenta. Biparental choriocarcinoma detected in a postpartum uterine sample is consistent with undetected intraplacental choriocarcinoma. Eutopic or ectopic androgenetic choriocarcinoma separate from a concurrent intrauterine placenta is not derived from intraplacental tumor and is consistent with either a form of dispermic twin gestation (molar-type choriocarcinoma and coexistent nonmolar fetus) or origin from an antecedent molar pregnancy. While fallopian tube tumors are usually gestational, tumors in other sites (ovary, pelvis) can be nongestational and should not be assumed to be metastatic from a regressed or occult intrauterine or intraplacental gestational tumor.


Subject(s)
Biomarkers, Tumor/genetics , Choriocarcinoma, Non-gestational/genetics , Choriocarcinoma/genetics , Ovarian Neoplasms/genetics , Uterine Neoplasms/genetics , Adolescent , Adult , Allelic Imbalance , Amelogenin/genetics , Choriocarcinoma/pathology , Choriocarcinoma/therapy , Choriocarcinoma, Non-gestational/pathology , Choriocarcinoma, Non-gestational/therapy , Chromosomes, Human, X , Chromosomes, Human, Y , Female , Genetic Predisposition to Disease , Heterozygote , Homozygote , Humans , Hydatidiform Mole/genetics , Hydatidiform Mole/pathology , Microsatellite Repeats , Middle Aged , Ovarian Neoplasms/pathology , Ovarian Neoplasms/therapy , Phenotype , Pregnancy , Prognosis , Uterine Neoplasms/pathology , Uterine Neoplasms/therapy , Young Adult
12.
World Neurosurg ; 95: 516-524.e1, 2016 Nov.
Article in English | MEDLINE | ID: mdl-27567571

ABSTRACT

BACKGROUND AND OBJECTIVE: Pediatric basal ganglia germ cell tumors (GCTs) represent a rare subset of tumors about which little is known. We aimed to summarize the clinical features and radiological findings of this special subgroup of GCTs. METHODS: From January 2010 to January 2015, 12 pediatric patients with basal ganglia GCTs were treated in our hospital. The clinical features, radiologic findings, diagnosis, treatment, and outcome of these patients were analyzed retrospectively. Our institutional diagnostic principle and treatment strategy of this disease were discussed. RESULTS: GCTs accounted for 25.5% of all the pediatric basal ganglia tumors treated in our hospital. There were 9 male and 3 female patients with a mean age of 11.5 ± 2.1 years. The most common symptom was progressive hemiparesis (n = 9, 75%). The radiologic findings showed that the lesions predominately located in caput of caudate nucleus (n = 9, 75.0%), followed by lenticular nucleus (n = 3, 25.0%). Hemiatrophy was commonly observed (n = 8, 66.7%). Eight patients were diagnosed as having germinomas, and 4 patients as having nongerminomatous germ cell tumors. During the follow-up period, preoperative neurologic dysfunctions improved in 7 patients and remained stable in 3. Two patients developed new onset of neurologic dysfunction after the treatment. Two patients suffered from tumor recurrence. CONCLUSIONS: GCTs are not as rare as considered in pediatric basal ganglia tumors. They bear some distinctive clinical and radiologic features, which can help with the accurate diagnosis and successful management of such tumors.


Subject(s)
Antineoplastic Combined Chemotherapy Protocols/therapeutic use , Basal Ganglia Diseases/therapy , Brain Neoplasms/therapy , Neoplasms, Germ Cell and Embryonal/therapy , Adolescent , Aftercare , Basal Ganglia Diseases/complications , Basal Ganglia Diseases/diagnostic imaging , Basal Ganglia Diseases/metabolism , Brain Neoplasms/complications , Brain Neoplasms/diagnostic imaging , Brain Neoplasms/metabolism , Carboplatin/administration & dosage , Carcinoma, Embryonal/complications , Carcinoma, Embryonal/diagnostic imaging , Carcinoma, Embryonal/metabolism , Carcinoma, Embryonal/therapy , Caudate Nucleus/diagnostic imaging , Caudate Nucleus/surgery , Child , Choriocarcinoma, Non-gestational/complications , Choriocarcinoma, Non-gestational/diagnostic imaging , Choriocarcinoma, Non-gestational/metabolism , Choriocarcinoma, Non-gestational/therapy , Chorionic Gonadotropin, beta Subunit, Human/metabolism , Cisplatin/administration & dosage , Cognitive Dysfunction/etiology , Corpus Striatum/diagnostic imaging , Corpus Striatum/surgery , Cranial Irradiation , Diffusion Tensor Imaging , Endodermal Sinus Tumor/complications , Endodermal Sinus Tumor/diagnostic imaging , Endodermal Sinus Tumor/metabolism , Endodermal Sinus Tumor/therapy , Etoposide/administration & dosage , Female , Germinoma/complications , Germinoma/diagnostic imaging , Germinoma/metabolism , Germinoma/therapy , Humans , Magnetic Resonance Imaging , Male , Neoadjuvant Therapy , Neoplasms, Germ Cell and Embryonal/complications , Neoplasms, Germ Cell and Embryonal/diagnostic imaging , Neoplasms, Germ Cell and Embryonal/metabolism , Neurosurgical Procedures , Paresis/etiology , Retrospective Studies , Second-Look Surgery , Seizures/etiology , Tomography, X-Ray Computed
13.
Am J Surg Pathol ; 38(1): 111-8, 2014 Jan.
Article in English | MEDLINE | ID: mdl-24145647

ABSTRACT

Although well recognized in the literature, the contemporary clinicopathologic data regarding choriocarcinoma (CC) as a pure or the predominant component of a testicular germ cell tumor (GCT) are limited. Herein, we present a series of pure CC and predominant CC in mixed GCT of the testis obtained from a single oncology institution. A comprehensive histologic review of 1010 orchiectomies from 1999 to 2011 yielded 6 (0.6%) pure CC and 9 (0.9%) mixed GCT cases with a predominant CC component. Patients' ages ranged from 20 to 39 years (median 29 y). All patients had markedly elevated serum ß-hCG levels (median 199,000 IU/mL) at presentation. All tumors were unilateral and involved the right (9/15) and left (6/15) testis. The mean tumor size was 6.5 cm (range, 1.5 to 8 cm). Histology was similar for pure CCs and the CC component of mixed GCTs. CC commonly showed expansile hemorrhagic nodular cysts surrounded by variable layers of neoplastic trophoblastic cells (mononucleated trophoblasts and syncytiotrophoblasts). The syncytiotrophoblasts usually covered columns of mononucleated trophoblasts and occasionally formed plexiform aggregates and pseudovillous protrusions. Immunohistochemical stains suggested a mixture of cytotrophoblasts (p63+, HPL_) and intermediate trophoblasts (p63-, HPL weak +/-) in the columns of mononucleated cells. In the 9 mixed GCTs, CC comprised 50% to 95% (7/9 were ≥80% CC) of the tumor; 7 were combined with 1, and 2 were combined with 2 other GCT components. The non-CC components included teratoma (5/9), seminoma (2/9), yolk sac tumor (2/9), and embryonal carcinoma (2/9). Lymphovascular invasion, spermatic cord invasion, and tunica vaginalis invasion were present in 15/15, 5/15, and 1/12 cases, respectively. In mixed GCTs, these locally aggressive features were attributed to the CC component, except in 1 tumor in which it was also exhibited by the embryonal carcinoma component. Lymphovascular invasion was multifocal to widespread in 73% of tumors. The stages of the 15 tumors were: pT2 (10), pT3 (5); NX (1), N1 (4), N2 (5), N3 (5); and M1a (2) and M1b (13). Distant organ metastasis mostly involved the lungs (11) and liver (10). Follow-up information was available in 14 patients, all of whom received cisplatin-based chemotherapy. All 6 pure CC patients were dead of disease (range, 6 to 14 mo, median 9.5 mo). Follow-up of 8 patients with predominant CC (range, 10 to 72 mo, median 27 mo) showed that 5 died of the disease, and 1 was alive with disease and 2 were alive with no evidence of disease at 60 and 72 months of follow-up, respectively; these latter 2 patients were the only ones with M1a disease on presentation. This series confirms the proclivity for high-stage presentation including presence of distant metastasis, hematogenous spread, and poor outcome of testicular CC. Mixed GCT with a predominant CC component has similar tendency for high-stage presentation, marked elevation of serum ß-hCG levels, and aggressive behavior compared with pure CC. This study also showed that distant metastasis by CC when only involving the lungs (M1a) may not be uniformly fatal with chemotherapy. The mononucleated trophoblastic columns in testicular CC appear to be a mixture of cytotrophoblasts and intermediate trophoblasts, similar to that described in gestational CC.


Subject(s)
Choriocarcinoma, Non-gestational/pathology , Neoplasms, Complex and Mixed/pathology , Testicular Neoplasms/pathology , Adult , Biomarkers, Tumor/analysis , Biopsy , Choriocarcinoma, Non-gestational/chemistry , Choriocarcinoma, Non-gestational/mortality , Choriocarcinoma, Non-gestational/secondary , Choriocarcinoma, Non-gestational/therapy , Humans , Immunohistochemistry , Lymphatic Metastasis , Male , Mexico , Neoplasm Invasiveness , Neoplasm Staging , Neoplasms, Complex and Mixed/chemistry , Neoplasms, Complex and Mixed/mortality , Neoplasms, Complex and Mixed/secondary , Neoplasms, Complex and Mixed/therapy , Testicular Neoplasms/chemistry , Testicular Neoplasms/mortality , Testicular Neoplasms/therapy , Treatment Outcome , Tumor Burden , Young Adult
14.
Vojnosanit Pregl ; 70(10): 968-71, 2013 Oct.
Article in English | MEDLINE | ID: mdl-24313181

ABSTRACT

INTRODUCTION: Gestational trophoblastic diseases (GTD) are a spectrum of tumors with a various of biological behavior and potential for metastases. It consists of hydatiform mole, invasive mole, choriocarcinoma and placental site trophoblastic tumor. Choriocarcinoma presents a very aggressive tumor with high malignant potential. CASE REPORT: We presented the two cases of choriocarcinoma with brain metastases. The first one was manifested by neurological deterioration as the first sign of metastasis, while the second patient had firstly metrorrhagia and in the further couse neurological disturbances that suggested the presence of brain tumor. In both cases we applied a combined treatment of surgery, chemotherapy and radiation therapy. Both patient survived with high quality of life. CONCLUSION: A successful outcome of brain metastases of choriocarcinoma was obtained by the use of a combined treatment of surgery, chemotherapy and radiation therapy. In cases of young women with brain metastases, gynecological malignancy should be always considered.


Subject(s)
Antineoplastic Agents/administration & dosage , Brain , Choriocarcinoma, Non-gestational , Neurosurgical Procedures/methods , Radiotherapy, Adjuvant/methods , Uterine Neoplasms , Adult , Brain/pathology , Brain/surgery , Brain Neoplasms/diagnostic imaging , Brain Neoplasms/physiopathology , Brain Neoplasms/secondary , Brain Neoplasms/therapy , Choriocarcinoma, Non-gestational/pathology , Choriocarcinoma, Non-gestational/physiopathology , Choriocarcinoma, Non-gestational/therapy , Combined Modality Therapy , Female , Humans , Radiography , Treatment Outcome , Uterine Neoplasms/pathology , Uterine Neoplasms/physiopathology , Uterine Neoplasms/therapy
15.
World J Gastroenterol ; 19(39): 6683-8, 2013 Oct 21.
Article in English | MEDLINE | ID: mdl-24151399

ABSTRACT

Primary colorectal choriocarcinoma is an extremely rare neoplasm and is usually associated with a poor prognosis. Only 13 cases of colorectal choriocarcinoma have previously been reported. There is no standard chemotherapeutic regimen for this tumor type. A 68-year-old man presented with melena and was diagnosed with sigmoid colonic adenocarcinoma with multiple liver metastases. He underwent a laparoscopic sigmoidectomy. Pathology revealed choriocarcinoma with a focal component of moderately differentiated adenocarcinoma of colon origin. Based on the collagen gel droplet-embedded culture drug sensitivity test (CD-DST) results, mFOLFOX6 and bevacizumab were administered, which suppressed aggressive tumor growth for 4 mo. The patient died 9 mo after the initial diagnosis. Our study results suggest that the standard chemotherapy regimen for colorectal cancer might have suppressive effects against primary colorectal choriocarcinoma. Moreover, CD-DST may provide, at least in part, therapeutic insight for the selection of appropriate antitumor agents for such patients.


Subject(s)
Adenocarcinoma/pathology , Choriocarcinoma, Non-gestational/pathology , Neoplasms, Complex and Mixed/pathology , Sigmoid Neoplasms/pathology , Adenocarcinoma/complications , Adenocarcinoma/secondary , Adenocarcinoma/therapy , Aged , Antibodies, Monoclonal, Humanized/administration & dosage , Antineoplastic Combined Chemotherapy Protocols/therapeutic use , Bevacizumab , Biopsy , Chemotherapy, Adjuvant , Choriocarcinoma, Non-gestational/complications , Choriocarcinoma, Non-gestational/secondary , Choriocarcinoma, Non-gestational/therapy , Colectomy , Colonoscopy , Disease Progression , Drug Screening Assays, Antitumor , Fatal Outcome , Humans , Liver Neoplasms/secondary , Male , Melena/etiology , Neoplasms, Complex and Mixed/complications , Neoplasms, Complex and Mixed/secondary , Neoplasms, Complex and Mixed/therapy , Sigmoid Neoplasms/complications , Sigmoid Neoplasms/therapy , Time Factors , Tomography, X-Ray Computed , Treatment Outcome
16.
World J Gastroenterol ; 19(31): 5187-94, 2013 Aug 21.
Article in English | MEDLINE | ID: mdl-23964157

ABSTRACT

Primary gastric choriocarcinoma is a rapidly growing neoplasm with an average survival of several months in untreated patients. Gastrectomy with lymph node dissection followed by chemotherapy is the treatment of choice. Regimens used for gastric adenocarcinoma are usually selected. However, median survival remains less than six months. In this case report, we describe a case of primary gastric choriocarcinoma with a clinical complete response to multidisciplinary treatment including surgery, chemotherapy, and radiofrequency ablation (RFA). The patient was originally referred for general malaise. Esophagogastroduodenoscopy demonstrated a large tumor occupying the fornix, and total gastrectomy with lymph node dissection was performed. Seven days later, multiple liver metastatic recurrences with high serum levels of beta-human chorionic gonadotropin (ß-hCG) were recognized. Chemotherapy with a gonadal choriocarcinoma regimen consisting of etoposide, methotrexate, actinomycin D, cyclophosphamide, and vincristine (EMA/CO), was initiated. After three cycles, serum ß-hCG decreased markedly and the tumors disappeared. Six months later, multiple lung metastatic recurrences were found. After one cycle of EMA/CO, only one nodule remained. Computed tomography-guided RFA was performed for this oligometastatic tumor. The patient has been alive with no evidence of disease for 10 years after the initial diagnosis. To the best of our knowledge, this patient with recurrent primary gastric choriocarcinoma has achieved the longest survival. The present case is the first report of choriocarcinoma metastatic to the lung successfully treated with RFA. From our retrospective analysis of recurrent or unresectable primary gastric choriocarcinoma, we propose that gonadal choriocarcinoma regimens can be considered as first-line for primary gastric choriocarcinoma.


Subject(s)
Antineoplastic Combined Chemotherapy Protocols/therapeutic use , Catheter Ablation , Choriocarcinoma, Non-gestational/therapy , Gastrectomy , Liver Neoplasms/therapy , Lung Neoplasms/therapy , Stomach Neoplasms/therapy , Aged , Biopsy , Choriocarcinoma, Non-gestational/secondary , Endoscopy, Digestive System , Female , Humans , Immunohistochemistry , Liver Neoplasms/secondary , Lung Neoplasms/secondary , Lymph Node Excision , Stomach Neoplasms/pathology , Time Factors , Tomography, X-Ray Computed , Treatment Outcome
19.
Int J Gynecol Pathol ; 29(6): 539-45, 2010 Nov.
Article in English | MEDLINE | ID: mdl-20881859

ABSTRACT

Ovarian epithelial tumors of nongerm cell origin with true choriocarcinomatous differentiation are rare. To date, there are only 5 documented cases in the literature. In the reported cases, the epithelial component was of mixed cell types or of mucinous differentiation. To the best of our knowledge, an ovarian carcinoma exclusively of clear cell differentiation coexisting with a pure choriocarcinoma has not been reported earlier. A 48-year-old postmenopausal woman was found to have a large pelvic mass with lung and liver metastases. Trucut biopsy of the mass showed a poorly differentiated carcinoma that was immunoreactive for CK7 and hCG. She received 6 cycles of neoadjuvant chemotherapy that included 3 cycles of etoposide/cisplatin and 3 cycles of paclitaxel/etoposide-paclitaxel/carboplatin (TE/TP) with partial response. Debulking surgery was carried out subsequently. Pathologic examination showed an ovarian clear cell carcinoma with a second component of choriocarcinoma in which the bilaminar growth pattern of cytotrophoblast and syncytiotrophoblasts was striking. Despite additional therapy, which included 2 cycles of TE/TP and 2 cycles of gemcitabine/taxotere, the disease progressed and the patient died 11 months postoperatively. This report showed that ovarian clear cell carcinoma with choriocarcinomatous differentiation is a highly aggressive tumor and has a very poor prognosis. Nonetheless, there may be a role for neoadjuvant chemotherapy that targets both the clear cell and the choriocarcinoma components to reduce the volume of the disease before debulking surgery.


Subject(s)
Adenocarcinoma, Clear Cell/pathology , Choriocarcinoma, Non-gestational/pathology , Ovarian Neoplasms/pathology , Adenocarcinoma, Clear Cell/therapy , Antineoplastic Agents/therapeutic use , Choriocarcinoma, Non-gestational/therapy , Combined Modality Therapy , Fatal Outcome , Female , Gynecologic Surgical Procedures , Humans , Ovarian Neoplasms/therapy
20.
Int J Gynecol Cancer ; 20(2): 299-302, 2010 Feb.
Article in English | MEDLINE | ID: mdl-20134273

ABSTRACT

INTRODUCTION: The objective of the study was to investigate the clinical characters, diagnosis, treatment, and prognosis of nongestational ovarian choriocarcinoma. METHODS: A retrospective analysis was done on 21 patients with nongestational ovarian choriocarcinoma treated in Peking Union Medical College Hospital from January 1985 to October 2008. All patients' conditions were diagnosed by histopathologic examination; in 3 of them, the diagnosis was confirmed by DNA polymorphism analysis at 12 short tandem repeat loci. RESULTS: Correct diagnosis was achieved in only 3 patients before initial treatment. All patients received standard multiple-drug combined chemotherapy and underwent an operation. The mean number of chemotherapy courses for each patient was 10. Of the 21 patients, 16 achieved complete remission, and 4 obtained partial remission; 1 died. In a median follow-up of 71.4 months, the 5-year overall survival rate was 79.4%. CONCLUSIONS: The early diagnosis of nongestational ovarian choriocarcinoma is expected to be improved. DNA polymorphism analysis is a useful tool in determining the origin of ovarian choriocarcinoma. The prognosis is optimistic if managed with standard multiple-drug chemotherapy combined with surgical treatment.


Subject(s)
Choriocarcinoma, Non-gestational/diagnosis , Ovarian Neoplasms/diagnosis , Adolescent , Adult , Child , China/epidemiology , Choriocarcinoma, Non-gestational/mortality , Choriocarcinoma, Non-gestational/therapy , Female , Humans , Middle Aged , Ovarian Neoplasms/mortality , Ovarian Neoplasms/therapy , Retrospective Studies , Treatment Outcome , Young Adult
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