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PubMed1984

Serial serum determination on alpha-fetoprotein as a marker of the effect of postoperative chemotherapy in ovarian endodermal sinus tumor.

Serial serum determinations of alpha-fetoprotein (alpha FP) as a tumor marker were carried out in the management of six patients with endodermal sinus tumor (EST). Histological examinations in all six patients revealed a typical EST pattern, and in one of them another germ cell tumor, a malignant teratoma, was also found. All patients were treated postoperatively with combination chemotherapy. The serum alpha FP concentrations before treatment, using radioimmunoassay, were abnormally high and ranged from 2500 to 100,000 ng/ml. One patient having Stage Ia neoplasm is living at 50 months after diagnosis with a normal alpha FP concentration and without clinical evidence of recurrence. The other five patients with Stage III disease died from 4 to 9 months after surgery, respectively, although the serum alpha FP in all these cases during chemotherapy decreased markedly but temporarily to a normal level (less than 20 ng/ml) or to 3000 ng/ml and was paralleled by a certain improvement in the conditions of the disease. These results suggest that serial serum determination of alpha FP may be useful as a marker and prognostic indicator of endodermal sinus tumor.

Adolescent↗
PubMed1996

Endodermal sinus tumor in children.

Malignant germ cell tumors account for about 3% of neoplasms in children, and endodermal sinus tumor (EST) is the most common histological subtype. The authors reviewed 22 years' experience (at their institution) in the management of 37 patients with this tumor. Fifteen of them (41%) had a sacrococcygeal primary, 10 had a testicular tumor (27%), 6 had an ovarian tumor (16%), 3 had a vaginal tumor (8%) and 3 had tumors at other sites (8%). Seven (19%) patients presented with metastatic disease, primarily pulmonary. The serum alpha-fetoprotein (AFP) level was elevated in all cases tested. The initial chemotherapy regimen included vincristine, actinomycin, cyclophosphamide (VAC), and Adriamycin (6 patients), but since 1985 the regimen has been changed to include cisplatin or Carboplatin, etoposide, and bleomycin (21 patients). Eight patients with testicular tumors initially were treated with surgical excision alone. Computed tomography results were a poor predictor of recurrence, but AFP surveillance was extremely sensitive. No second-look operation detected residual tumor in the absence of AFP elevation. Initial relapse for all patients tended to occur early (within 2 years), locally, and often with pulmonary metastases. Although historically the prognosis for patients with EST has been poor, the overall 2-year survival rate in this series was 70%. The best prognosis was among the children who had a testicular primary tumor (survival rate, 100%). The 2-year survival rate for patients with ovarian tumors was 67%; for those with sacrococcygeal primaries it was 60%. These results suggest that the prognosis for children with EST has improved significantly over the past decade. Contributing factors include therapy based on cisplatin, etoposide, and bleomycin, and relapse surveillance with serial AFP determinations. Second-look procedures should be reserved for patients who have an increasing level of serum AFP, suspicious computed tomography findings, and no obvious evidence of metastatic disease.

Adolescent↗
PubMed1996

Deletion of 1p36 in childhood endodermal sinus tumors by two-color fluorescence in situ hybridization: a pediatric oncology group study.

Childhood endodermal sinus tumors (CESTs) are a unique category of germ cell tumors involving the testis and extragonadal region in children less than 4 years of age. Recent studies of CEST have shown recurrent cytogenetic abnormalities involving the short arm of chromosome 1, most commonly, a deletion of distal 1p. Experience with neuroblastomas has shown that cytogenetic analyses may underestimate the frequency of 1p deletion. To determine the frequency of deletion of Ip in CEST and to verify that 1p is, in fact, deleted and not translocated, we analyzed ten tumors by two-color fluorescence in situ hybridization on single-cell suspensions of interphase nuclei by using a cosmid probe from the PITSLRE kinase (p58) locus (previously mapped to 1p36) cohybridized with plasmid probe pUC1.77 (which recognizes the 1q heterochromatic region) to determine the copy number of chromosome 1. Eight of the ten tumors examined showed evidence of deletion of 1p36. Five of the eight tumors exhibited multiple subdones, and all subdones showed deletion of at least one copy of 1p36, indicating that the deletion probably occurred before the development of chromosome 1 aneusomy. We conclude that deletions of the short arm of chromosome 1, specifically 1p36, do occur in CEST and probably occur at a, higher incidence than that found in neuroblastoma Further studies are needed to determine the degree of overlap of the common area of deletion in CEST with that of neuroblastoma and to determine whether 1p deletion in CEST has prognostic significance.

Child, Preschool↗
PubMedDate not supplied

Disseminated peritoneal tuberculosis mimicking advanced-stage endodermal sinus tumor: a case report.

It is well known that peritoneal tuberculosis may mimic advanced-stage epithelial ovarian carcinoma because of similar clinical, radiologic, and laboratory findings. However, disseminated peritoneal tuberculosis mimicking advanced-stage endodermal sinus tumor (ESS) has not been reported previously. An 18-year-old nulliparous woman came with the complaint of pelvic pain and weight loss. Imaging studies demonstrated that she had multiple peritoneal implants and left adnexial mass. Also, laboratory studies showed elevated CA125 and alpha fetoprotein levels suggesting an initial diagnosis of ESS. However, intraoperative frozen section examination showed caseous necrosis, and she was diagnosed as having disseminated peritoneal tuberculosis. Two months after the initial exploration, the patient required liver transplantation because of hepatic failure due to widespread hepatic involvement of the tuberculosis. Concomitant peritoneal and hepatic involvement of tuberculosis may cause false elevation of multiple tumor markers of gynecological cancers and may lead to misdiagnosis and mismanagement of patients. Elevation of these markers should be carefully investigated especially in premenopausal women. To our knowledge, this is the first reported case of peritoneal tuberculosis misdiagnosed as endodermal sinus tumor.

Adolescent↗
PubMed1980

[Effect of anti-human alpha-fetoprotein serum on human endodermal sinus tumor transplanted in nude mice (author's transl)].

The effects of horse anti-human alpha-fetoprotein (AFP) immunoglobulin (IgG) on growth, serum AFP concentration and histopathology of human endodermal sinus tumor serially xeno-transplanted in nude mice were examined. Anti-AFP IgG given in amounts sufficient to neutralize the serum AFP secreted by the tumor moderately reduced tumor size and significantly decreased the serum AFP levels of treated nude mice. On histopathologic examination, spindle shaped tumor cells of reticular pattern often fell into the state of necrosis. But little change was observed on the cells consisting of both solid patterns and Schiller-Duval bodies. With a growth rate of re-transplanted tissues respectively, which were obtained from both antibody administrated and control tumors, no disparity was found between two groups. Indeed this result is preliminary report, but inhibitory effects of anti-AFP IgG against tumor growth and AFP producing ability of endodermal sinus tumor xeno-transplanted in nude mouse is hopeful to apply these data on clinical cancer immunochemotherapy.

Animals↗
PubMedDate not supplied

Successful chemotherapeutic decompression of primary endodermal sinus tumor presenting with severe spinal cord compression.

Management of spinal cord compression from a primary paraspinal endodermal sinus tumor (EST) is described. A 17-month-old child presented for treatment with near-complete paraplegia secondary to spinal cord compression from a primary paraspinal EST. The child was treated with cisplatin-based chemotherapy without laminectomy or radiation therapy. Rapid resolution of symptoms was observed. The child had an excellent tumor response and complete neurologic recovery with no sequelae. Chemotherapy alone is an alternative to laminectomy or radiation therapy in the management of epidural cord compression from EST, even when the cord compression is severe.

Endodermal Sinus Tumor↗
PubMedDate not supplied

Effusion cytology of endodermal sinus tumor of the colon. Report of a case.

A three-year-old boy presented with a peritoneal effusion due to an occult endodermal sinus tumor in the ascending colon. Cytologic examination of the ascitic fluid revealed clusters of round plump cells that had large hyperchromatic nuclei, finely stippled chromatin, multiple prominent nucleoli and vacuolated or mucin-containing cytoplasm, features suggesting an adenocarcinoma. The colonic primary was discovered during emergency laparotomy performed due to suspected acute hemoperitoneum. Endodermal sinus tumor should be included in the differential diagnosis when cytologic features reminiscent of adenocarcinoma are encountered in a fluid specimen from a child, especially if there is a history of a gonadal or extragonadal childhood neoplasm.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed1985

Endodermal sinus tumor of the vagina and cervix.

This report describes six patients with endodermal sinus tumor of the vagina and cervix, a polypoid friable tumor whose clinical presentation in girls younger than age 3 years simulates the presentation of sarcoma botryoides. In four of the six patients, the referring diagnosis was sarcoma botryoides. Five patients were treated with excisional surgery, and all six with chemotherapy. One patient with pulmonary metastases maintained a complete clinical response to vincristine, actinomycin-D, and cyclophosphamide (VAC) for 11 months. This is the first report of such a response. One patient with a vaginal lesion remains clinically free of disease 2 years after local excision and 18 months of VAC chemotherapy. This is the first report of apparently successful therapy that allowed retention of childbearing potential. Four of the six patients have been disease-free from 2 to 23 years. After examining the world literature, it is concluded that a combination of chemotherapy and surgery offers a reasonable prospect of cure with a minimum of serious side effects.

Adolescent↗
PubMedDate not supplied

Malignant sacrococcygeal yolk sac (endodermal sinus) tumor.

We report a 2 1/2 years old female child who presented with Malignant Sacrococcygeal Yolk sac (Endodermal Sinus) tumor as presacral mass with ulceration of overlying skin. Her ultrasonography (USG)abdomen revealed a huge presecral mass with irregular margins extending in to the pelvis, pushing the rectum antero-laterally. CT scanning of abdomen confirmed USG finding with out enlargement of retroperitoneal lymph nodes and no involvement of the liver. Serum AFP was abnormally elevated. The tumor was excised along with Coccyx in-toto through posterior approach dividing the sacrum and 3-Drugs (VAC) Chemotherapy started postoperatively i.e. 2-weeks after surgery. Here we discuss our experience with this extremely rare malignant extra-gonadal germ-cell tumor and review of literature.

Child, Preschool↗
PubMed1980

Endodermal sinus tumor (yolk sac tumor), seminoma and hemangiopericytoma. Rare primary mediastinal tumors. A report on the course of the disease in nine patients.

The pathological and clinical features of primary mediastinal hemangiopericytoma, seminoma, and endodermal sinus tumor are presented and discussed. These tumors were found in 3 patients each out of 898 cases of mediastinal tumor. Primary thoracic hemagiopericytomas are rare. The diagnosis can be determined by histology only, but nothing can be said about the malignancy of the tumor. Tumor recurrence is relatively frequent. Two patients were radically operated, and they are living today 4 and 4 1/2 years later, without evidence of recurrence. The third patient with clinically clearly malignant hemangiopericytoma was inoperable. This patient died 6 months after diagnosis. The anterior mediastinum is a predilection site for extragonadal germ cell tumors. Histologically there is no difference between this tumor and the gonadal seminoma. This tumor is well known to be radiosensitive. All 3 male patients were inoperable and received radiotherapy. One patient died after 4 months, the other 2 are living one and 3 years later, respectively, without complaints or evidence of recurrence. Only 12 cases of primary mediastinal endodermal sinus tumors have been reported in the literature, we observed 3 more such tumors. The highly malignant alpha-fetoprotein producing tumor occurs exclusively in young males. There is, as yet, no clear-cut form of treatment. One of our patients died after 2 weeks, another received chemotherapy and died a few months after diagnosis, the third received chemotherapy and irradiation and is living today, 20 months after discovery of the tumor.

Adult↗
PubMedDate not supplied

Integrated 18F-fluorodeoxyglucose positron emission tomography and computerized tomography in endodermal sinus tumor.

Application of an in-line positron emission tomography and computerized tomography (PET-CT) in endodermal sinus tumor (EST) is described in this study. CASE 1: A young female with massive ascites postovarian mass resection had elevated alpha-fetoprotein (AFP) serum levels. Following a positive PET-CT study with increased (18)F-fluorodeoxyglucose (FDG) uptake, a CT-guided core biopsy of a peritoneal mass was performed. EST was diagnosed histologically. The patient was disease free after chemotherapy. Follow-up PET-CT was negative in keeping with no viable tumor tissue. CASE 2: A large pelvic mass diagnosed histologically as primarily EST was removed in a teenage patient with elevated AFP levels. PET-CT showed diffuse abdominal spread of FDG uptake, suggesting extensive peritoneal seeding. The patient was disease free after chemotherapy. Follow-up PET-CT was negative. EST is an FDG-avid tumor. PET-CT delineated the prechemotherapy tumor extent adequately ruled out the presence of residual tumor after a successful treatment.

Adolescent↗
PubMed2005

Vaginal endodermal sinus tumor.

Malignant germ cell tumors are rare tumors of childhood accounting less than 3% of pediatric malignancies, and endodermal sinus tumor (EST) is the most common histological subtype. The vagina is an extremely rare site for germ cell tumors (GCT). A one-year female was admitted with history of bleeding per vagina. She had pallor and a mass was palpable anteriorly on rectal examination. Magnetic Resonance Imaging (MRI) showed a tumor mass at the junction of cervix and vagina and biopsy was suggestive of a vaginal EST. The serum alpha fetoprotein (AFP) was elevated. She underwent vaginohysterectomy. The tumor was excised in toto and patient received four courses of cisplatin, etoposide and bleomycin. At one year of follow up, patient was symptom free and serum AFP remained at normal level. EST of the vagina is a rare, highly malignant GCT that exclusively involves children less than 3 years of age. It is both locally aggressive and capable of metastasis. Even though more conservative surgery is advised to maintain sexual and reproductive function, at times, radical surgery is mandatory depending on the infiltration of the tumor to the surrounding structures. Simple tumor excision is not sufficient, as residual cells induce recurrence and make chemotherapy ineffective. The serum AFP level is a useful marker for diagnosis and monitoring the recurrence of vaginal EST in infants.

Endodermal Sinus Tumor↗
PubMed1976

Embryonal carcinoma of the ovary: a clinicopathologic entity distinct from endodermal sinus tumor resembling embryonal carcinoma of the adult testis.

The clinical and pathologic features of 15 examples of a hitherto undescribed germ cell tumor of the ovary are delineated. This tumor resembles the embryonal carcinoma of the adult testis and may be distinguished from the endodermal sinus tumor on the basis of its histologic and immunohistochemical characteristics. An indirect immunoperoxidase method for the localization of human chorionic gonadotropin (HCG) and alpha-fetoprotein (AFP) was done on formalin-fixed paraffin-embedded tissue from 10 neoplasms; HCG was present in all 10 neoplasms, and AFP was found in seven. HCG was indentified only in cells resembling syncytiotrophoblast, whereas AFP was present only in mononuclear embryonal cells, indicating that the two protein antigens were localized in different cell types. Abnormal hormonal manifestations, consisting of precocious puberty, irregular bleeding, amenorrhea, or hirsutism, were present in nine (60%) of the patients. The actuarial survival for the entire group was 39%; for those with stage I tumors, 50%. We are proposing the term "embryonal carcinoma" for this neoplasm in order to distinuish it from the more common endodermal sinus tumor of the ovary and to emphasize the histologic similarity to embryonal carcinoma of the adult testis.

Adolescent↗
PubMed1980

Immunohistochemical demonstration of a hitherto undescribed localization of hemoglobin A and F in endodermal cells of normal human yolk sac and endodermal sinus tumor.

In this study of 4 human yolk sacs, the presence of hemoglobin A and F (HbA and HbF) is demonstrated for the first time in epithelial cells (type 1) and erythroid-like cells (type 2) in the endodermal layer by immunoperoxidase technique. Our findings strongly support the hypothesis previously proposed that the red blood cells formed in the yolk sac are of endodermal origin. Tumor with yolk sac differentiation (8 endodermal sinus tumors and 1 embryonal carcinoma with vitelline areas) similarly showed HbA and HbF localisation in endodermal cells. None of 59 germ cell tumors of other types contained these hemoglobins in the neoplastic cells.

Endoderm↗
PubMed1998

An endodermal sinus tumor arising from a mature cystic teratoma in the retroperitoneum in a child: is a mature teratoma a premalignant condition?

We herein report an 18-month-old girl who presented with a retroperitoneal mature teratoma that later developed into an endodermal sinus tumor. Her symptoms included an expanding abdominal girth. The serum levels of alpha-fetoprotein were moderately elevated. The excised specimen was a benign mature cystic retroperitoneal teratoma measuring 18 x 11 x 8 cm in size. A hemorrhagic focus measuring 4 cm in diameter was present, which proved to be the focus of an endodermal sinus tumor histologically. The retroperitoneal mature teratoma observed in this case showed malignant germ-cell differentiation. The above case is thought to be extremely rare, but these findings also suggest the possibility of a mature teratoma presenting as a premalignant condition. The sequence of "mature teratoma--germ-cell malignancy" is thus considered to represent a new potential pattern of early phase of carcinogenesis in teratomas.

Chorionic Gonadotropin↗
PubMed1991

An endodermal sinus tumor in the cerebellopontine angle.

Immunohistochemical and ultrastructural findings in a primary intracranial endodermal sinus tumor are reported in this paper. The tumor cells exhibited AFP, CEA and anti-alpha-1-trypsin positive immunoreactivity immunocytochemically. Aggregates of electron-dense material in the extra- and intracellular spaces and amorphous basement membrane-like substance were seen extracellularly by electron microscopy. The clinicopathological, immunocytochemical and ultrastructural features were consistent with the criteria for primary intracranial sinus tumor.

Cerebellar Neoplasms↗