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PubMedDate not supplied

Endodermal sinus tumor of the vulva: successfully treated with high-dose chemotherapy.

Endodermal sinus tumors (EST) of the lower female genital tract are uncommon malignancies. Most of these tumors are found in the vagina or in the cervix. Only seven cases of EST involving the vulva are reported. We report an eighth case of vulvar EST. The patient recurred and was successfully treated with high-dose chemotherapy and peripheral blood cell transplantation. Because of their location, an early diagnosis of vulvar EST should be possible. Resection of the lesion by using a modified radical vulvectomy followed by cisplatin-based chemotherapy seems the best treatment for these tumors today. Our patient was initially treated with the help of local surgery and adjuvant chemotherapy consisting of three courses of etoposide and cisplatin. Sixteen months after the first diagnosis, pleural and lung metastases were diagnosed. Second-line chemotherapy followed by high-dose chemotherapy with autologous peripheral blood stem cell support was administered after resection of the lung metastases and biopsy of the pleural metastases. There is no evidence of disease 40 months after the diagnosis of the lung and pleural metastases, and 56 months after the primary diagnosis.

Adult↗
PubMed2001

Deletion mapping of 6q21-26 and frequency of 1p36 deletion in childhood endodermal sinus tumors by microsatellite analysis.

The most common malignant germ cell tumor of early childhood is the endodermal sinus tumor (CEST), also known as yolk sac tumor. Previous cytogenetic studies of CEST have demonstrated recurrent deletion of distal regions of chromosomes 1p and 6q. Studies utilizing comparative genomic hybridization have likewise demonstrated loss of distal 6q, however these studies show discrepant data concerning chromosome 1 abnormalities. This study analyses 18 CESTs for loss of heterozygosity (LOH) of distal chromosome 6q utilizing 17 microsatellite markers and 13 tumors were analysed for LOH of distal 1p using two microsatellite markers. LOH of 6q was found in 13/18 tumors (72 %). This data confirms that loss of genetic material on 6q is one of the most common abnormalities in CESTs and narrows the region of loss, enabling candidate tumor suppressor genes to be identified and analysed. In addition, LOH of 1p36 was identified in five of 11 informative tumors, clarifying prior conflicting data and confirming that 1p deletion is a common event in CESTs.

Child, Preschool↗
PubMed1985

Endodermal sinus tumor and embryonal carcinoma of the ovary in children.

The clinicopathological features of 26 pure endodermal sinus tumors (EST) and for embryonal carcinomas of the ovary occurring in children under age 15 years in Britain over the period 1962-1978 are described. Endodermal sinus tumor has shown a significant increase (P less than 0.001) over the 17-year period considered. Compared to controls, EST and embryonal carcinoma have not shown an association with the use of hormones or tranquilizers during pregnancy, with radiological investigations, or with infections in pregnancy though the numbers of cases studied were very small. The histological and immunohistological features of EST and embryonal carcinoma in children do not differ substantially from the descriptions of these tumors in adolescents and adults. Using the FIGO staging classification 23% were Stage I tumors; 3% were Stage II; 70% were Stage III; and 3% were Stage IV neoplasms. The 5-year actuarial survival rate was 39% among 26 cases of EST and 1 of the 4 cases of embryonal carcinoma also survived for 5 years. Clinical stage at diagnosis was the only indicator of prognosis, but the introduction of polychemotherapy (vincristine, actinomycin-D, cyclophosphamide/cis-platinum, vincristine, bleomycin) resulted in long-term survival in 7 of 7 cases of EST (2 Stage I and 5 Stage III) and 1 of 2 cases of embryonal carcinoma (Stage III). Radiotherapy appears to have little role to play in the treatment of pure EST or embryonal carcinoma.

Actuarial Analysis↗
PubMed1978

Primary yolk-sac (endodermal sinus) tumor of the prostate.

A primary yolk-sac (endodermal sinus) tumor of the prostate occurring in a 51-year-old man is described, and the difficulties associated with classification of this tumor are discussed. Yolk-sac tumors have been reported to arise in several extragonadal sites, but to our knowledge this is the first documented case of a germinal cell tumor occurring the the prostate. This tumor in extragonadal sites carries a grave prognosis. Despite radical surgery, radiotherapy, and chemotherapy, this patient died 10 months after the tumor was diagnosed.

Dysgerminoma↗
PubMed1985

Endodermal sinus tumor of the vagina. The role of primary chemotherapy.

Endodermal sinus tumor of the vagina, a rare pediatric malignancy, was managed with primary chemotherapy (vincristine, dactinomycin, cyclophosphamide [VAC]) followed by local excision without radical surgery or pelvic irradiation. Treatment-related complications were acceptable, and there is no evidence of disease 30 months after discontinuing all therapy (50 months after diagnosis). As a treatment option, primary chemotherapy is viewed as a highly attractive alternative modality, preserving potential reproductive and sexual function in long-term survivors, and appears to be the treatment of choice.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed1989

Aggressive chemotherapy in endodermal sinus tumor.

Twenty-eight patients with pure or predominantly endodermal sinus tumor were studied for their clinical behavior, relation with serum alpha-fetoprotein (AFP), and response to intensive postoperative combination chemotherapy. Eight percent of the patients were younger than 20 years of age. One patient was a pseudohermaphrodite. Serum AFP was estimated in 17 patients and was elevated in all. Seven patients were treated with adjuvant chemotherapy following surgery with a cisplatin-containing combination. Five patients (71%) are alive and continue to remain free of disease, with a median survival of 28 months after treatment and an overall median survival of 34 months. Two patients treated with adjuvant chemotherapy experienced early recurrence of the disease, which was detected with a rise in serum AFP before the clinical manifestation of recurrence.

Adolescent↗
PubMed1985

Cisplatin, vinblastine, and bleomycin therapy of yolk sac (endodermal sinus) tumor of the ovary.

Five patients with yolk sac (endodermal sinus) tumor of the ovary were treated with cisplatin, vinblastine, and bleomycin combination therapy (PVB). Four of five achieved a complete remission and remain free from disease 24 to 53 months from start of PVB therapy. One patient did not respond well to PVB and died 11 months after start of PVB therapy. One patient who was treated with PVB after unilateral salpingo-oophorectomy has delivered a normal term infant. Serum alpha-fetoprotein levels were monitored in all patients during and after therapy. Serum alpha-fetoprotein was correlated with clinical course.

Adolescent↗
PubMed1984

Intrapleural endodermal sinus tumor arising from the diaphragm.

We report a primary endodermal sinus tumor (EST) of the right hemidiaphragm, presenting as a right-sided hemothorax in an 18-month-old female. Four operations were performed, the last three being indicated solely by an elevated level of serum alpha-fetoprotein (AFP). Cyclic chemotherapy with varying drug combinations together with irradiation therapy were not effective in controlling the disease. To our knowledge, this is the first instance of a primary EST arising from the diaphragm.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed1993

Retrospective review of 41 patients with endodermal sinus tumor of the ovary.

Forty-one patients with endodermal sinus tumors of the ovary (EST) (23: pure EST, nine: EST with dysgerminoma; and nine: EST with immature teratoma) were treated with cytoreductive surgery, with or without subsequent adjuvant chemotherapy including VAC or PVB. Clinical staging of these patients showed 23 in stage I, six in stage II and 12 in stage III and IV. The outcome was good in the patients with stage I or II disease, with 5- and 10-year survival rates of around 80%. In contrast, all except two patients with stage III or IV disease died within 2 years. Aggressive surgery did not change the outcome of patients with stage I disease. In addition, postoperative chemotherapy was necessary, but the choice of vincristine, actinomycin-D and cyclophosphamide (VAC) or cis-platin, vinblastine and bleomycin (PVB) did not make any difference. Based on the results of the present study, VAC or PVB chemotherapy, after cytoreductive surgery, is essential for cure and conservative surgery followed by VAC or PVB regimen should be recommended for young patients.

Journal Article↗
PubMed1984

Florida cluster of five children with endodermal sinus tumors. Possible environmental risk.

An unusual case clustering of endodermal sinus tumors in Jacksonville, Fla., is described, together with a variety of local environmental risk factors. Proximity to high-tension electrical power lines was observed, but chance aggregation of familially disposed persons or coincidental environmental factors not identified could also be responsible. However, this cluster is too remarkable to go unreported and is presented here as a preliminary report for those interested in the epidemiology of rare tumors, in an attempt to probe this peculiar episode of cancer. Further investigation of cancer clusters is encouraged, with suggestions to maintain surveillance for these types of clusters in comparably affected areas.

Adolescent↗
PubMed1988

Successful treatment of pure endodermal sinus tumors in adult men.

Seven adult men with pure endodermal sinus tumors (EST) were treated with cyclical combination chemotherapy Cytoxan (cyclophosphamide; Bristol-Myers Company, Evansville, IL), Adriamycin (doxorubicin; Adria Laboratories, Columbus, OH), and cisplatin/vinblastine and bleomycin (CISCAII/VBIV) and surgery at the University of Texas M.D. Anderson Hospital and Tumor Institute at Houston from 1978 through 1985. Six tumors were of extragonadal origin (four anterior mediastinum, one pelvic, one prostate), and one was of gonadal origin with retroperitoneal metastasis. All patients presented with advanced local disease and a relative absence of distant metastasis. Alpha-fetoprotein (AFP) levels were elevated in six patients (median, 4,400 ng/mL; range, 2,580 to 31,200 ng/mL). Six patients achieved a complete remission (CR): one with chemotherapy alone, one with initial surgery followed by chemotherapy, and four with chemotherapy followed by consolidative surgery. The remaining patient died of progressive disease. Of the six patients who achieved a CR, five are alive with no evidence of disease (+17, +23, +34, +43, +59 months); one patient developed recurrent disease at 6 months after completion of therapy and is currently undergoing salvage chemotherapy. Of the four patients who underwent postchemotherapy surgery, three were operated on for a marker-negative stable mass; in these patients, no viable tumor was found at pathologic review. The remaining patient underwent surgery for a stable mass with a persistent elevation in AFP levels. He was found to have 95% necrosis with 5% viable tumor and remains disease free without further therapy. The observed changes in AFP levels correlated with regression and progression of tumor; a normal AFP was consistent with a CR, and elevation was consistent with residual tumor. These seven patients demonstrate that when adult men with EST are treated aggressively with combination chemotherapy and surgery, high cure rates can be achieved.

Adult↗
PubMed1980

Endodermal sinus tumor arising in the endometrium.

A case of primary endodermal sinus tumor (EST) of the endometrium in a 28-year-old woman is described. EST has been reported to arise in several extragonadal sites, but to the authors' knowledge this is the first documented instance of origin in the endometrium. The histologic study is verified by the detection of alpha-fetoprotein in the tumor by an immunoperoxidase-peroxidase-antiperoxidase technique reported here. Displaced germinal cells, abnormal ovum, and residual fetal tissues are considered as possible origins of the neoplasia.

Adult↗
PubMed1981

Endodermal sinus tumor of the ovary with virilization. Light- and electron-microscopic study.

A case of endodermal sinus tumor of the ovary associated with hirsutism and increased testosterone production is described. Electron-microscopic examination revealed accumulation of basement membrane-like material and other ultrastructural features consistent with endodermal sinus tumor. Groups of polyhedral cells associated with the tumor were found to contain abundant lipid material. These luteinized stromal cells were evidently responsible for the endocrine manifestations.

Adult↗
PubMed1993

Non-midline endodermal sinus tumor in the head and neck region: a case report.

Germ cell tumors, in particular teratomas, are some of the most commonly found tumors in childhood. Six percent of all germ cell tumors are located in the head and neck region. Endodermal sinus tumors (yolk sac tumors) of the head and neck, exclusive of the central nervous system, are rare. This study reports a 20-month-old girl with a mass of 5 x 5 cm on the left temporal area and with bone destruction on CT. The histopathological examination of the excised mass revealed an endodermal sinus tumor. The serum AFP and the LDH levels were elevated at diagnosis. The patient was treated by the BEP protocol (bleomycin, etoposide, and cisplatin), six cycles every three weeks. Partial response was attained after the first and complete response after the fourth cycle. The patient, who is being followed up, has been in remission for five months.

Endodermal Sinus Tumor↗
PubMed2007

Endodermal sinus tumor of the vagina.

Malignant germ cell tumors of the vagina represent a critical site for local treatment. We report on our experience with two vaginal endodermal sinus tumors. Treatment with PEB regimen induced both tumor regression and alpha fetoprotein normalization. One patient had a residual lesion that was biopsied twice and no viable tumor cells were ever found. Both patients remained alive and disease-free, without having had surgical procedures except for biopsies. Platinum-based chemotherapy is able to achieve complete remissions and should be considered for vaginal endodermal sinus tumors.

Antineoplastic Combined Chemotherapy Protocols↗
PubMed1993

[A case of endodermal sinus tumor in the basal ganglia associated with Down's syndrome].

A case of Down's syndrome accompanied by endodermal sinus tumor of the left basal ganglia is reported. She was referred to us because of right hemiparesis. A CT scan showed a calcified mass in the left basal ganglia, which was well enhanced on contrast CT. Emergency removal of the tumor was performed because of its rapid growth, intratumoral hemorrhage, and acute hydrocephalus. The histological diagnosis established after surgery was endodermal sinus tumor. Alphafetoprotein-positive cells were seen in the resected tumor by light microscopy. Postoperative irradiation was very effective, and the high level of serum alphafetoprotein decreased during the course of irradiation. The residual tumor, enhanced on contrast CT, was treated by administration of cisplatin and etoposide after irradiation. Cases of malignant tumors other than leukemia, which are associated with Down's syndrome, are extremely rate. To our knowledge, including three cases of germ cell tumor, there have been only nine cases of brain tumors associated with Down's syndrome reported previously.

Adolescent↗