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A patient with a pineal endodermal sinus tumor is presented who was successfully treated by a combination of surgery, adjuvant chemotherapy, and craniospinal irradiation. Two years after diagnosis, he is free of any disease. A review of the literature shows that such an outcome is very unusual. A multidisciplinary treatment is recommended for this rare tumor, using chemotherapy as adjuvant treatment.
Endodermal sinus tumor (EST) of the ovary is extremely rare and little information exists about therapy and the role of second-look laparotomy in the management of this entity. A case of EST of the ovary in a 21 year old woman is reported. She received conservative surgery and six courses of combination therapy consisting of Vincristine, Actinomycin D and Cyclophosphamide before second-look laparotomy. Due to progression of the disease second-line polychemotherapy with Vinblastine, Bleomycin and Cisplatin was administered. This new regimen reduced the alpha-fetoprotein to normal levels although the patient was not free of disease on second-look laparotomy. Precise guidelines for the management of this disease, especially in advanced stages, are still lacking.
An unusual case of endodermal sinus tumor (EST) of the ovary at an extragonadal site-vulva, in an unmarried female of 25 yr is reported. The patient presented only with a vulval swelling on the right side without any other signs or symptoms. The internal genital organs mainly the ovaries were normal.
An own case of a primary mediastinal endodermal sinus tumour (yolk sac tumour) is presented that was transformed under therapy into an AFP-negative process and occurred combined with an arcus aortae dexter. This is followed by a description of the clinical and diagnostic patterns of signs and symptoms and of the treatment of this very rare dysontogenetic tumour.
An asymptomatic 11-year-old Japanese boy with an endodermal sinus (yolk sac) tumor of the anterior mediastinum was successfully treated with cisplatinum-based combination chemotherapy and subsequent surgical excision. The patient is alive and in good health 60 months from diagnosis.
Basement membrane procollagen biosynthesis was studied in organ cultures of embryonic rat parietal yolk sac endoderm by following [14C]proline incorporation into nondialyzable proteins. After reduction with 2-mercaptoethanol the 14C-proteins synthesized were characterized by agarose gel filtration and disc electrophoresis in the presence of sodium dodecyl sulfate. The labeled procollagen was identified by its content of hydroxy[14C]proline, its sensitivity to digestion with bacterial collagenase, and its resistance to digestion with pepsin. In cultures which were continuously labeled for periods from 6 hours to 4 days, the pro-alpha chains consistently eluted as a single peak with an apparent molecular weight of 160,000. After pepsin digestion the resultant alpha chains had an apparent molecular weight between 125,000 and 140,000. This suggests that basement membrane procollagen either contains non-triple helical pepsin-resistant regions or a triple helical region which is larger than the corresponding region of interstitial procollagen. Two experiments were performed to determine whether the chains of newly synthesized basement membrane procollagen were cleaved to a smaller molecular species. In the first, the hydroxylation and secretion of procollagen were blocked with alpha, alpha'-dipyridyl, and the resulting intracellular chains of basement membrane protocollagen were found to co-elute with fully hydroxylated and secreted pro-alpha chains. In the second, cultures were labeled for 1 day and chased for 3 days with unlabeled medium. Autoradiography had shown that most of the label was chased into new basement membrane. Agarose chromotography showed that after 3-day chase the pro-alpha chains still eluted with an apparent molecular weight of 160,000. Thus, the data indicated that basement membrane procollagen was deposited in new basement membrane without undergoing a time-dependent extracellular conversion.
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.
The case of a child affected at birth sacrococcygeal teratoma is reported. Twenty-eight months following surgical resection, the tumor relapsed locally and liver metastases occurred. A biopsy of the sacrococcygeal mass was performed and histologic examination proved it to be an endodermal sinus (yolk sac) tumor. Chemotherapy consisting of etoposide and high-dose carboplatin was started. When a second operation was performed the mass had disappeared and the macroscopically involved areas of liver were removed. Histologic examination demonstrated that the complete necrosis of the tumor was partially replaced by a fibrous scar tissue. The child underwent adjuvant chemotherapy and now is in complete remission fourteen months after the last operation.
A case of an extragonadal endodermal sinus tumour in a three-year old boy is presented. Complete resection of the retroperitoneal tumour was possible after initial chemotherapy. Combined radio-chemotherapy after tumour removal led to complete remission now lasting for more than 5 years. There was unilateral renal atrophy and diverticulum of the stomach as treatment related complications. The favourable prognosis of this tumour due to intensified treatment is discussed in relation to treatment sequelae. The interdisciplinary approach in treatment of cancer in children is emphasized.
The combination of cisplatin, vinblastine, and bleomycin was administered to 13 previously untreated patients with pure endodermal sinus tumor of the ovary. Twelve had unilateral salpingo-oophorectomy as initial surgery. Among 11 women whose alpha-fetoprotein (AFP) levels returned to normal after cisplatin, vinblastine, and bleomycin therapy, nine are alive with no evidence of disease after periods of 20 months to six years and two months. In two patients, AFP levels returned to normal only after the introduction of different combination chemotherapies. Hematologic toxicity was moderate. Pulmonary toxicity due to bleomycin occurred in two cases. All menstruating patients resumed normal menstrual function after the interruption of chemotherapy. Two patients delivered normal term infants.
We studied 3 cases of a variant of endodermal sinus tumor (EST) or yolk sac tumor (YST). Two tumors originated in the ovary and one in the testis. The patients had very high levels of serum alpha-fetoprotein (AFP). All three tumors had a characteristic histologic appearance and were composed of acinar structures lined by columnar epithelium with large, immature nuclei. Immunohistochemistry and electron microscopy, including freeze-fracture studies, confirmed that this unusual tumor is one with exclusive intestinal differentiation. We feel that this unique tumor is a pure EST (YST) with exclusive intestinal differentiation.
52 cases of endodermal sinus tumors (EST) are reported and 1224 cases from the Chinese and foreign literature are reviewed. This tumor, occurring usually in the gonads and rarely at extragonadal sites, comprised 10-17.1% of the former. It is located on or along the mid-line of the body including the orbit and other organs. In this paper, there are two cases with the tumor located at the orifice of urethra and one case between the pubic bone and the urinary bladder which has never been reported. EST often occurs in people under the age of 35. EST in the gonadal sites is divided into infant-childhood and adult groups. In the former group the prognosis is better than that of the adult group when the tumor is located in the testis as well as those in the vagina. There is sex and age difference for some EST in extragonadal sites. Those in the mediastinum almost always occur in young males. Diagnosis is easily made by pathology and by serum alpha-fetal protein (SAFP). Pathologically it is divided into two types: simple EST and EST element mixed with other germ cell tumors which give the same prognosis. The authors suggest that SAFP be done for any patients with teratomata in order to avoid misdiagnosis. SAFP is not only diagnostic but also prognostic by monitering postoperative course. EST is a highly malignant tumor, rapidly growing and poor in prognosis. Recent reports show that operated patients supplemented by chemotherapy give much better results than by radiotherapy.
An ovarian endodermal sinus tumor was found in a patient with a 15-week intrauterine gestation. She received conservative surgery and six courses of combination chemotherapy from 16 weeks' gestation, and delivered a normal term infant at 37 weeks' gestation. Two other courses of combination chemotherapy including cisplatin were added. No residual tumor was found at subsequent exploration, and she is alive 33 months after the original diagnosis without any evidence of recurrence. We believe this is the first reported case of maternal survival past 2 years and the earliest chemotherapy done in the early second trimester of pregnancy without resulting in fetal abnormalities.
Discussed is a case of a stage IV pure endodermal sinus tumor (EST) of the ovary that was treated with a PVB protocol following cytoreductive surgery. The serum alpha-fetoprotein (AFP) dosage before treatment was 83,546 ng/ml, and a complete clinical response was achieved with the disappearance of pleural effusions, ascites, and normalization of the serum AFP. A second-look laparotomy was substituted by serial AFP determinations and the patient remained free of disease for two years. Recently, complete remissions and long term survivals of patients with a stage I-II disease have been reported, though a prolonged or a complete remission of a stage IV EST of the ovary is still a rarity.
Reported is a rare case of a 2.5cm endodermal sinus tumor (EST) that was included within a 11 x 10.5 cm serous cystadenoma. A 24-year-old female suddenly complained of abdominal pain caused by having undergone an intestinal torsion of a left ovarian tumor. After a unilateral salpingo-oophorectomy the serum AFP was 219.8 ng/ml. The patient then received two cycles of cis-platinum, vinblastine and bleomycin combination chemotherapy. After one cycle, the serum AFP decreased to a normal level and the woman has remained free of disease 6 months after the discovery of her tumor. In this case, fortunately, this torsion of a serous cystadenoma enabled us to find the EST at a very early stage.
An endodermal sinus tumour of the anterior mediastinum is described in a 19-year-old man. This very rare condition is reported for the first time in the Caribbean literature.
Eight cases of mediastinal cysts of endodermic and celomatic origin were operated on at Istituto di Chirurgia Generale e Cardiovascolare, University of Milano: two were bronchogenic cysts, two enteric and four pericardial. Embryology, morphologic presentation, clinical feature and diagnostic aspects are discussed for each group of cysts. The most modern techniques, radiological or not, available for the preoperative diagnosis are illustrated and the indication to surgical treatment of these benign mediastinal lesions is discussed.
Three cases of Endodermal Sinus Tumor are presented and its difference from tumors of the mesonephroma type and associations with the embryoma are pointed out. The three cases were "pure" types and the patients died within 5 months in spite of surgical treatment and chemotherapy. Autopsy showed extensive involvement of peritoneum, diaphragm and pleura.