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PubMed1991

[Germ-cell tumors of the testis in childhood].

Germinative-cell tumors are the most common tumors of the testis in children. For a period of 15 years (1975-1989) the authors have observed 12 children with these tumors. Leading symptoms in all children was a gradual painless enlargement of one testis; the symptomatic hydrocele may delay the correct diagnosis for several months. In young children the mothers noticed the testicular changes earlier, while in older children the diagnosis was delayed. Three quarters of the tumors were in the left testis, seldom in the right and still more seldom bilaterally. Diagnostic methods were applied mostly when metastases or recurrence were suspected; most common were computer tomography and lymphography. The most common benign testicular tumors were the mature teratomas and malignant--endodermal sinus tumors. The modern operative approach to these patients is emphasized: radical orchiectomy with pre- and postoperative determination of AMP levels, periodic control computer tomography and echography of the retroperitoneum and thorax with 2-year observation in stage I and modified lymphadenectomy in stage II. As a result of the complex surgical and oncologic therapy, the late results were good in 7 children, 2 died and 3 were lost from control.

Child↗
PubMed1983

Ovarian Sertoli-Leydig cell tumors with a retiform pattern: a problem in histopathologic diagnosis. A report of 25 cases.

A review of 232 cases of ovarian Sertoli-Leydig cell tumors yielded 25 that contained a prominent component with a retiform pattern. A lack of awareness of this pattern, which has not been described adequately in the literature, resulted in frequent initial misinterpretation of these tumors as neoplasms of other types, including endodermal sinus tumor (seven cases) and serous adenocarcinoma (four cases). The patients ranged in age from 2 to 39 years. Three of them (12%) presented because of androgenic manifestations; most of the remainder complained of abdominal swelling or pain, but one of them was amenorrheic and hirsute and three had amenorrhea, suggesting androgen secretion by the tumor. All the tumors were Stage I; one was bilateral. Their average maximum dimension was 16 cm and they were typically cystic or both cystic and solid. In eight tumors the cysts were lined by papillae, which were edematous in five cases. On microscopic examination the retiform pattern, which predominated in 14 of the 25 tumors, was characterized by an irregular network of elongated, often slit-like tubules and cysts, which often contained papillae and resembled the rete testis. Follow-up of 21 patients from 6 months to 17 years (average 4 years) revealed that 16 of them were alive and free of disease; five had died of tumor from 6 months to 17 years postoperatively.

Adolescent↗
PubMed1992

Endodermal sinus (yolk sac) tumor of the parotid gland: a case report.

Malignant salivary gland neoplasms in children are rare, most common being mucoepidermoid carcinoma, adenoid cystic carcinoma, acinic cell carcinoma and adenocarcinoma. Most germ-cell neoplasms of head and neck in children are teratomas. The authors report a case of endodermal sinus tumor (EST) of the parotid gland in a 2-year-old girl, which recurred after chemotherapy. The role of alpha-fetoprotein (AFP) serum level as a helpful marker in differential diagnosis and in evaluating tumor progression is underlined.

Child, Preschool↗
PubMed1999

Treatment of nondysgerminomatous ovarian germ cell tumors: an analysis of 69 cases.

BACKGROUND: Combination chemotherapy has dramatically improved the prognosis of patients with nondysgerminomatous ovarian germ cell tumors (NDOGCT). However, guidelines are needed for the identification of patients at risk of relapse. METHODS: The authors performed a retrospective analysis of women with NDOGCT managed during the period 1970-1994 at the Royal Marsden Hospital and other hospitals of the London Gynaecological Oncology Group. RESULTS: Sixty-nine women were included; their median follow-up was 5.7 years (minimum, 12 months). The median age was 21 years (range, 4-44 years), with a histology of immature teratoma (IT) for 17 patients, endodermal sinus tumor (EST) for 20 patients, and mixed tumors for 32 patients. Thirty-five patients (51%) had Stage I disease. Nine patients with Stage I tumors were observed without further therapy (six with IT and three with mixed tumors), and one relapsed. Seven patients received non-platinum-based chemotherapy, and four relapsed. A total of 52 patients were treated with platinum-based chemotherapy, with relapse free and overall survival rates of 87% (95% confidence interval [CI], 73-93%) and 84% (95% CI, 70-91%), respectively. Of these patients, relapse was seen in 0 of 9 IT patients, 1 of 25 patients with mixed tumors, and 6 of 18 EST patients. With alpha-fetoprotein (AFP) > 1000 kU/L, relapse was seen in 6 of 18 patients compared with 1 of 33 relapses with lower AFP levels. In multivariate analysis, including all patients who received chemotherapy, AFP >1000 kU/L (P = 0.001) and non-platinum-based chemotherapy (P = 0.005) were associated with relapse. When only patients given platinum-based treatment were considered, EST histology (P = 0.003) and AFP >1000 kU/L (P = 0.003) were associated with relapse in univariate analysis; however, these factors were linked. No malignant tumor was found at second-look surgery performed on 24 patients. Of 26 women assessable for fertility, 24 subsequently recommenced regular menstrual function, and 11 patients had pregnancies. CONCLUSIONS: Platinum-based chemotherapy has been confirmed to be effective in the management of patients with NDOGCT. Relapses were principally seen among patients with AFP >1000 kU/L or pure EST histology. Efforts to improve outcome need to focus on patients with EST, whereas less intensive management strategies may be appropriate for some patients with IT.

Adolescent↗
PubMed1993

Rectal adenocarcinoma with germ cell elements treated with chemotherapy.

A patient with adenocarcinoma of the rectum containing germ cell elements in the form of endodermal sinus tumor and choriocarcinoma is described, believed to be the first such tumor reported in the literature. The serum levels of alpha-fetoprotein and human chorionic gonadotropin were markedly elevated. The source of the markers was localized to the neoplastic tissue by the immunoperoxidase technique. The patient developed extensive hepatic metastases thought to be due to choriocarcinoma for which she received chemotherapy. One wk after commencing treatment a massive fatal hepatic hemorrhage occurred.

Adenocarcinoma↗
PubMed1998

Long-term outcome for infants and children with sacrococcygeal teratoma: a report from the Childrens Cancer Group.

BACKGROUND/PURPOSE: Sacrococcygeal teratomas (SCT) are a relatively uncommon tumor affecting neonates, infants, and children. This study was designed to determine the effect of therapy on the long-term outcome of neonates and children with sacrococcygeal teratomas (SCT). METHODS: The authors conducted a retrospective review of children with SCT treated at 15 Childrens Cancer Group institutions from 1972 to 1994. RESULTS: One hundred twenty-six children presented with SCT diagnosed prenatally (n = 32), at birth (n = 79), or later in infancy (n = 15). For neonates, complete resection was performed except in two babies with lethal associated defects. All others (n = 15) underwent resection at the age of diagnosis. Six had a sacral mass identified at birth but had delayed surgery (1.5 to 11 months) and of these, two were malignant. Resection was via sacral (n = 96) or abdominosacral (n = 28) approach. Histology showed mature teratoma (MT, 69%), immature teratoma (IT, 20%), or endodermal sinus tumor (EST, 11%) at presentation. Seven neonates (5.6%) died of perioperative complications, whereas the remaining 117 were available for long-term follow-up. Between 6 and 34 months postresection, recurrent disease developed in 9 of 80 MT patients (11%) followed-up for a mean of 5 years. Recurrent disease was MT (n = 2) and EST (n = 7). The recurrent EST patients were treated with adjuvant chemotherapy. Six are alive with mean follow-up of 114 months, whereas one with metastatic disease was lost to follow-up. Recurrence (MT) developed in only 1 of 24 IT patients, and all are alive and well at mean follow-up of 39 months. Patients presenting with EST (n = 13) underwent excision, with two dying from non-EST causes. Six EST patients received no chemotherapy, with two of the six (33%) experiencing recurrence within 11 months and both disease free after salvage chemotherapy. The remaining five EST patients received adjuvant chemotherapy; four are alive and one died of metastatic disease. Of the 18 EST patients followed-up after resection (presentation, 11, recurrent teratoma, 7), 16 (89%) are free of disease with a mean follow-up of 91 months. CONCLUSIONS: (1) Benign teratomas have a significant recurrence rate mandating close follow-up for more than 3 years. (2) Surgical resection alone is adequate therapy for nonmetastatic malignant tumors. (3) Survival for malignant lesions with metastases is excellent with modern chemotherapy.

Child, Preschool↗
PubMed1987

Ovarian Sertoli-Leydig cell tumor (androblastoma) with retiform pattern. A clinicopathologic study.

The clinicopathologic findings in nine patients with ovarian Sertoli-Leydig cell tumor with retiform pattern are described. The patients ranged in age from 11 months to 23 years; and seven patients were 12 years of age or younger. The most frequent presenting sign was the finding of an abdominal mass. This was associated with pain in five patients. In three patients the pain was severe due to torsion, causing an acute abdominal emergency. Slight virilization was observed in one patient only. Two patients had elevated serum alphafetoprotein (AFP), which correlated well with disease activity. The remaining patients had normal serum AFP. All the tumors were unilateral. At laparotomy the tumor was intact in six patients and ruptured in three. The tumors ranged from 8 to 22 cm, were round or oval, and cystic or solid and cystic. Eight tumors were in FIGO Stage I, and one was associated with abdominal metastases and was Stage III. Histologically, the retiform component varied from moderate to predominant in eight of the nine cases. In two tumors a heterologous component composed of striated muscle was also present. Three patients developed metastases. Two of the patients died 11 months and 2 years after diagnosis and the third patient was lost to follow-up with evidence of disease 2 years after diagnosis. The remaining six patients were well and disease-free for periods of 8 months to 6 years. The majority of these tumors were misinterpreted as serous papillary cystadenocarcinoma or endodermal sinus tumor, which are more malignant neoplasms requiring different therapy. This further underlines the importance of recognizing this histopathologic entity.

Adolescent↗
PubMed1998

Evaluation and management of pediatric solid ovarian tumors.

Solid ovarian tumors are uncommon in the pediatric population, but when they occur, they are a major source of anxiety for the patient and her family. The pediatric surgeon will be relied on to diagnose these tumors because they usually present as abdominal pain with a mass. The diagnostic evaluation consists of a carefully obtained history and physical examination, ultrasound examination, serum assay for tumor markers, and further radiographic evaluation as indicated. Two thirds of malignant tumors in children are germ cell tumors, and most of these are dysgerminomas or endodermal sinus tumors. A multimodal, team-oriented approach to therapy is crucial. Reproductive organ-sparing surgery with salpingo-oophorectomy, ascites sampling, nodule biopsy, omentectomy, and contralateral ovarian biopsy as indicated, may be curative for stage I tumors; more advanced or highly aggressive tumors should be treated with cytoreduction surgery and will require platinum-based chemotherapy. Postinduction surgery is indicated for progressive or recurrent disease. One third of tumors are physiologically active stromal tumors that often become apparent because of hormonal effects. Epithelial tumors, common in the adult, are uncommon in children and are of mild to moderate malignant potential. Other miscellaneous tumors and benign lesions are less common.

Carcinoma, Embryonal↗
PubMed1985

Anterior mediastinal endodermal sinus (yolk sac) tumor in a female infant.

The first known case of primary anterior mediastinal endodermal sinus (yolk sac) tumor in a female patient, occurring in a 20-month-old infant, is reported. The child presented with cough, fever, and listlessness. Chest x-ray revealed a right anterior mediastinal mass. At thoracotomy a large anterior mediastinal tumor extending from the neck to the diaphragm was found, and was almost totally resected. Microscopically, the tumor displayed many of the histologic patterns observed in EST. Other neoplastic germ cell elements were not identified. The ultrastructural and immunohistochemical findings further confirmed the diagnosis. Serum alpha-fetoprotein (AFP) level, determined during surgery, was elevated to 65,200 ng/ml, whereas serum beta-human chorionic gonadotropin level was normal. Postoperatively, combination chemotherapy consisting of vinblastine, bleomycin, cisplatin, dactinomycin, cyclophosphamide, and doxorubicin was administered with a maintenance program. After 18 weeks on this regimen all the findings were normal, including serum AFP level. The child is well and disease-free 25 months after diagnosis.

Antineoplastic Combined Chemotherapy Protocols↗
PubMedDate not supplied

Experience with pineal region tumors.

The results are reported of a retrospective review of the presentation and outcome of 43 pineal region tumors treated from 1982 to 1996, including 20 identified tumors: 5 germinomas, 8 teratomas, 2 embryonal carcinomas, 1 endodermal sinus tumor, 2 pineocytomas and 2 pineoblastomas. Of the 43 tumors reviewed, 36 were located in the pineal region, 5 in the suprasellar, and 2 in both the pineal and suprasellar regions. Twenty patients underwent surgical resection: total in 6 and partial in 10, while only a biopsy was taken in 4 cases. Fifteen patients were managed on the basis of serum CSF tumor markers and radiation response. Twenty-three patients with germinomas received radiotherapy (RT) and had a 5-year survival rate of 87%. Fifteen patients with non-germinomatous germ cell tumors received RT and chemotherapy following direct surgery, and 5 died (mortality rate of 33.3%). The overall survival rate of the 43 patients with pineal tumors was 79.1% (34/43) and the death rate was 20.9% (9/43). It is now recognized that the wide variety of tumor types found in the pineal region necessitates different modes of treatment, and improved microsurgical and stereotactic surgical techniques have made mortality and morbidity rates acceptably low. Because the radiation response and CSF cytology are not enough to determine optimum treatment, a tissue diagnosis should be obtained in all patients.

Adolescent↗
PubMed1995

Genital bleeding in premenarcheal children.

OBJECTIVE: The earlier occurrence of secondary sexual development and the advance in diagnostic techniques prompted us to review our recent experience with genital bleeding in childhood. METHOD: We analyzed data for all patients aged less than 10 years who were referred to Gifu University School of Medicine-affiliated hospitals from 1984 through 1993. RESULTS: From a total of 330 girls, 62 (approximately 20%) complained of genital bleeding. The patients were distributed equally between 0 and 10 years of age. In 46 of 62 patients (74%), bleeding resulted from a local lesion of the vagina: 28 due to vulvovaginitis, six to urethral prolapse, six to trauma, three to foreign bodies and three to vaginal tumors. Malignant vaginal tumors were seen in two patients: sarcoma botryoides and endodermal sinus tumor. Both were diagnosed by histopathological examination of an exophytic vaginal tumor, with several weeks' interval from the initial episode of bleeding to diagnosis. The remaining 16 patients (26%), with no demonstrable local lesions, bled as a result of some form of precocious puberty. In six patients, precocious puberty was secondary to a hormonally active ovarian tumor, five of these patients also experiencing advanced breast development. Routine ultrasound and hormonal evaluations promptly detected the presence of tumor. None of three patients with idiopathic precocious puberty presented with secondary sexual development. No specific etiology was established in seven patients. CONCLUSION: Genital bleeding alerts us to the possibility, though rare, of a genital tract tumor. Prompt and precise detection may lead to cure and preservation of future fertility.

Age Distribution↗
PubMed1996

Diagnosis and management of malignant germ cell ovarian tumors in young females.

Germ cell tumors (GCT) compose most of the preadolescent malignant ovarian tumors; dysgerminoma being the most common (48%), followed by endodermal sinus tumor, immature teratoma, mixed GCT and embryonal carcinoma. The percentage of malignant epithelial ovarian tumors rises with increasing age, while that of the GCT tumors declines. Of all tumor markers discussed, only AFP and hCG are being routinely monitored. Their most effective use is in monitoring response to therapy and detecting recurrences early. The current therapeutic regimens are presented, among them bleomycin, etoposide and platinol (BEP) and other new regimens; their influence on the patients' fertility is discussed. Further improvement in the prognosis of these young patients will hopefully follow development of new surgical and chemotherapeutic approaches.

Adolescent↗
PubMed1991

Ovarian tumors in the second decade of life.

A study of 28 adolescents aged 10-20 years admitted with ovarian tumors during a 5-year period revealed that 75% had malignant ovarian tumors, with germ cell tumors being most frequent. The high incidence of malignancy was due to our hospital being a referral centre. One case of mixed germ cell tumor (mainly choriocarcinoma) presented with precocious puberty, and one case of arrhenoblastoma showed signs of virilization. Tumor markers were found useful in diagnosing choriocarcinoma and endodermal sinus tumor. Amongst the malignant ovarian tumors, the best prognosis was noted with dysgerminomas. Tumors were found more frequently on the right side. Optimal surgery at initial laparotomy, followed by adjuvant chemotherapy or radiotherapy were found to offer the best change of cure.

Adolescent↗
PubMed1984

Primary malignant mediastinal tumors.

Thirty-eight patients with primary malignant mediastinal tumors of all cell types are the basis for this review. Eleven of these patients had germ cell tumors. Five germ cell tumors were seminomas, two were malignant teratomas, and two were endodermal sinus tumors. Mean survival for all patients with germ cell tumors was 3.3 years. Eight children had surgical excision of mediastinal neuroblastomas, and all but 1 are alive for a mean survival of 6.7 years. Seven patients had lymphoproliferative disorders; 6 of these patients had nodular sclerosing Hodgkin's disease, and 1 had lymphoblastic (thymic) lymphoma. Mean survival was 5.1 years. There were five carcinomas of various cell types and one angiopericytoma. None of the patients with these lesions survived more than 2 years. Four patients had thymoma with an average survival of 3.7 years. Two patients had carcinoid tumors of thymic origin; neither survived more than 1 year. In 1972, we reported 5-year disease-free survival of 26% in a series of patients with primary mediastinal tumors. Our experience since 1970 shows current survival of 47.3% and 5-year disease-free survival of 34.2%. We use combined methods of therapy, including aggressive surgical resection, combination chemotherapy, and often mediastinal irradiation for most types of mediastinal tumors. Primary mediastinal malignancies should be treated aggressively using a multidisciplinary approach, since many of these tumors are curable.

Adolescent↗
PubMed1986

Two murine monoclonal antibodies against human lung cancer-associated antigens.

Two murine monoclonal antibodies, MAb 8 (immunoglobulin G3 kappa) and MAb 15 (immunoglobulin G1 kappa), were produced after immunization with TKB-2, a variant cell line of human small cell carcinoma of the lung. In enzyme-linked immunosorbent assay, these antibodies reacted with four major types of lung cancer cell lines and various extrapulmonary tumor cell lines. Immunohistological study, however, showed highly specific binding to lung cancers; MAb 8 bound to 68% of 65 lung cancers, and MAb 15 bound to 72% of them. Interestingly, both antibodies were more reactive with non-small cell than small cell lung cancers and bound most frequently to large cell carcinoma. Most extrapulmonary tumor tissues were negative in staining with a few exceptions; endodermal sinus tumor (two of two) was positive to both antibodies, breast carcinoma (one of five) to MAb 8, gastric carcinoma (one of three), and malignant melanoma (one of one) to MAb 15. Cross-reactions with normal tissues were limited; MAb 8 reacted with adult and fetal lung, and MAb 15 with esophagus and renal tubules. MAb 8 recognized a Mr 48,000 glycoprotein antigen (carbohydrates as its epitope), and MAb 15 recognized two proteins (Mr 85,000 and 45,000) (peptides as their epitopes). These two antibodies, detecting novel antigens extensively associated with and highly specific to lung cancers, are potentially useful for the study of lung cancer.

Animals↗
PubMed1986

[Importance of immunohistochemistry for neuro-oncology. IV. Distribution model of beta human chorionic gonadotropins in intracranial germ cell tumors].

beta-Human choriogonadotropic hormone (beta-HCG) is considered a good marker for trophoblastic differentiation of germ cell tumors. 34 primary intracranial germ cell tumors (15 germinomas, 6 mature teratomas, 1 embryonal carcinoma, 2 endodermal sinus tumors and 10 mixed germ cell tumors) were immunohistochemically evaluated for the presence of beta-HCG positive cells. In 8 of 15 germinomas and 6 of 10 mixed germ cell tumors beta-HCG cells were demonstrable. In the germinomas such cells included both syncytiotrophoblastic and mononuclear cells which histologically did not correspond to the cytotrophoblast. In one case the patient had exhibited a precocious puberty. Of the 6 beta-HCG positive mixed germ cell tumors, two contained elements of choriocarcinoma. In the cytotrophoblasts of the choriocarcinoma regions, beta-HCG was only sparsely demonstrable. Both of these patients had manifest precocious puberty clinically. The advantage of immunohistochemical demonstration of the beta-HCG compared to conventional histology is in the definite identification of trophoblastic differentiation, in particular the exact recognition of the choriocarcinoma segments, which can be critical for the prognosis. Demonstration of isolated syncytiotrophoblasts and beta-HCG positive mononuclear cells in the seminomas is of no prognostic significance and is primarily of theoretical interest.

Adolescent↗
PubMed1984

[Human B-HCG and AFP as biological markers in germinal testicular tumors].

Alpha-fetoprotein (AFP) and the beta subunit of human chorionic gonadotropin (BHCG) were determined in 64 cases of testicular germinal tumors. Pure seminomas do not produce either AFP or BHCG, endodermal sinus tumors always produce AFP and never BHCG. Testicular germinal tumors may produce BHCG (44.4 per 100) and AFP (33.3 per 100), one or both at a time. There is a significant correlation between the amount of BHCG and AFP on the one hand, and tumor size and tumor dissemination on the other. When therapy is effective, BHCG and AFP become negative, while they remain positive in case of incomplete tumor removal or turn again to positive in case of relapse.

Adult↗