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1. Suarez CR, Bertolone SJ, Raj AB, Coventry S: Second malignant neoplasms in childhood acute lymphoblastic leukemia: primitive neuroectodermal tumor of the chest wall with germline p53 mutation as a second malignant neoplasm. Am J Hematol; 2004 May;76(1):52-6
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  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Second malignant neoplasms in childhood acute lymphoblastic leukemia: primitive neuroectodermal tumor of the chest wall with germline p53 mutation as a second malignant neoplasm.
  • Second malignant neoplasm (SMNs) are a devastating sequelae observed on these children, with an estimated cumulative risk of 2-3.3% fifteen years after diagnosis.
  • Primitive neuroectodermal tumor of bone (PNET) is rarely observed as a SMN following treatment of childhood ALL.
  • The authors described the occurrence of a chest wall PNET of the bone at the site of a central line placement associated with both germ-line and tumor cell p53 mutation in a 8-year-old boy 1 year after completing therapy for standard risk ALL.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Neoplasms, Second Primary / genetics. Neuroectodermal Tumors, Primitive, Peripheral / genetics. Precursor Cell Lymphoblastic Leukemia-Lymphoma / drug therapy. Thoracic Wall / pathology. Tumor Suppressor Protein p53 / genetics
  • [MeSH-minor] Child, Preschool. Combined Modality Therapy. Humans. Male. Mutation. Treatment Outcome

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  • [Copyright] Copyright 2004 Wiley-Liss, Inc.
  • (PMID = 15114597.001).
  • [ISSN] 0361-8609
  • [Journal-full-title] American journal of hematology
  • [ISO-abbreviation] Am. J. Hematol.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / Tumor Suppressor Protein p53
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2. Fujita K, Tsujimura A, Miyagawa Y, Kiuchi H, Matsuoka Y, Takao T, Takada S, Nonomura N, Okuyama A: Isolation of germ cells from leukemia and lymphoma cells in a human in vitro model: potential clinical application for restoring human fertility after anticancer therapy. Cancer Res; 2006 Dec 1;66(23):11166-71
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  • [Title] Isolation of germ cells from leukemia and lymphoma cells in a human in vitro model: potential clinical application for restoring human fertility after anticancer therapy.
  • More than 70% of patients survive childhood cancer, but chemotherapy and radiation therapy may cause irreversible impairment of spermatogenesis.
  • To treat infertility secondary to anticancer treatment for childhood cancer, we have developed a procedure to isolate germ cells from leukemic mice by fluorescence-activated cell sorting with two surface markers, and transplantation of isolated germ cells successfully restored fertility without inducing leukemia.
  • In the present study, we analyzed human germ cells and human malignant cells, including five leukemia cell lines and three lymphoma cell lines, by fluorescence-activated cell sorting with antibodies against MHC class I and CD45.
  • In the high forward scatter and low side scatter region, no malignant cells were found in the MHC class I-negative and CD45-negative fraction (the germ cell fraction), with the exception of K562 cells.
  • A total of 39.2% of the germ cells were found in the germ cell fraction.
  • A total of 1.45% of K562 cells were found in the germ cell fraction.
  • Treatment with IFNgamma induced the expression of MHC class I on K562 cells but not on germ cells and made it possible to isolate germ cells from K562 cells.
  • In conclusion, we isolated human germ cells from malignant cells with two surface markers after treatment with IFNgamma.
  • [MeSH-major] Cell Separation / methods. Flow Cytometry / methods. Germ Cells / cytology
  • [MeSH-minor] Antigens, CD45 / genetics. Antigens, CD45 / metabolism. Cell Line, Tumor. Drug-Related Side Effects and Adverse Reactions. Fertility / drug effects. Fertility / radiation effects. HL-60 Cells. HLA-A Antigens / genetics. HLA-A Antigens / metabolism. HLA-B Antigens / genetics. HLA-B Antigens / metabolism. HLA-C Antigens / genetics. HLA-C Antigens / metabolism. Humans. Immunohistochemistry. Infertility / etiology. Infertility / therapy. Jurkat Cells. K562 Cells. Leukemia / genetics. Leukemia / metabolism. Leukemia / pathology. Lymphoma / genetics. Lymphoma / metabolism. Lymphoma / pathology. Male. Radiotherapy / adverse effects. Reverse Transcriptase Polymerase Chain Reaction. Testis / cytology. Testis / metabolism. U937 Cells

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  • (PMID = 17145860.001).
  • [ISSN] 0008-5472
  • [Journal-full-title] Cancer research
  • [ISO-abbreviation] Cancer Res.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
  • [Chemical-registry-number] 0 / HLA-A Antigens; 0 / HLA-B Antigens; 0 / HLA-C Antigens; EC 3.1.3.48 / Antigens, CD45
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3. Kim E, Bae TS, Kwon Y, Kim TH, Chung KW, Kim SW, Ro J, Lee ES: Primary malignant teratoma with a primitive neuroectodermal tumor component in thyroid gland: a case report. J Korean Med Sci; 2007 Jun;22(3):568-71
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  • [Title] Primary malignant teratoma with a primitive neuroectodermal tumor component in thyroid gland: a case report.
  • Teratomas comprise the most common extragonadal germ cell tumors in childhood.
  • However, the adult cases reported are mostly malignant and commonly arise in the thyroid.
  • Pathologic examination revealed it to be malignant teratoma composed of primitive neuroepithelial tissue with primitive neural tubes and loose myxoid to fibrous immature mesenchymal stroma.
  • The patient underwent extensive evaluation of the thyroid gland with computed tomography (CT) scan and positron emission tomography (PET) scan, which revealed no evidence of metastatic disease.
  • She underwent total thyroidectomy with bilateral modified radical neck dissection, intensive chemotherapy and radiotherapy.
  • This is the first case, to our knowledge, of malignant thyroid teratoma with a exuberant primitive neuroectodermal tumor component in Korea.
  • [MeSH-minor] Adult. Female. Head and Neck Neoplasms / pathology. Humans. Neoplasm Metastasis. Positron-Emission Tomography / methods. Thyroid Diseases / diagnosis. Thyroidectomy. Tomography, X-Ray Computed

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  • (PMID = 17596674.001).
  • [ISSN] 1011-8934
  • [Journal-full-title] Journal of Korean medical science
  • [ISO-abbreviation] J. Korean Med. Sci.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Korea (South)
  • [Other-IDs] NLM/ PMC2693658
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4. Lo Curto M, Lumia F, Alaggio R, Cecchetto G, Almasio P, Indolfi P, Siracusa F, Bagnulo S, De Bernardi B, De Laurentis T, Di Cataldo A, Tamaro P: Malignant germ cell tumors in childhood: results of the first Italian cooperative study "TCG 91". Med Pediatr Oncol; 2003 Nov;41(5):417-25
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  • [Title] Malignant germ cell tumors in childhood: results of the first Italian cooperative study "TCG 91".
  • BACKGROUND AND AIMS: About 20% of patients with germ cell tumor (GCT) are still resistant to therapy.
  • The site of the primary tumor was gonadal in 59, extragonadal in 36.
  • The treatment was surgery alone in 31; surgery plus radiotherapy in 1; chemotherapy +/- surgery in 63.
  • Post-chemotherapy resection in 19 (10 complete, 9 partial).
  • The chemotherapy regimen was carboplatin 400 mg/m2/day on days 1, 2; etoposide 150 mg/m2/day on days 1, 2; ifosfamide 1,500 mg/m2/day on days 21, 22; dactinomycin 1.5 mg/m2/day on day 21; vincristine 1.5 mg/m2/day on day 21.
  • Three patients died because of toxicity and two non-responders (to primary chemotherapy), died of progression; among the remaining 90 patients 20 relapsed, 9 are in second remission, 2 are alive with disease, and 9 died of disease progression (one from progression and intracranial hemorrhage).
  • All the pts who had complete resection of the primary tumor at diagnosis or at delayed surgery, remained in remission.
  • CONCLUSIONS: Multivariate analysis showed that the primary site of tumor was the only independent prognostic factor for survival and EFS.
  • [MeSH-major] Germinoma / pathology. Germinoma / therapy. Ovarian Neoplasms / pathology. Ovarian Neoplasms / therapy. Testicular Neoplasms / pathology. Testicular Neoplasms / therapy
  • [MeSH-minor] Adolescent. Age Distribution. Child. Child, Preschool. Cohort Studies. Combined Modality Therapy. Confidence Intervals. Female. Humans. Incidence. Italy / epidemiology. Male. Multivariate Analysis. Neoplasm Staging. Probability. Prognosis. Retrospective Studies. Risk Assessment. Sex Distribution. Survival Analysis. Treatment Outcome

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  • [Copyright] Copyright 2003 Wiley-Liss, Inc.
  • (PMID = 14515380.001).
  • [ISSN] 0098-1532
  • [Journal-full-title] Medical and pediatric oncology
  • [ISO-abbreviation] Med. Pediatr. Oncol.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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5. De Backer A, Madern GC, Hakvoort-Cammel FG, Oosterhuis JW, Hazebroek FW: Mediastinal germ cell tumors: clinical aspects and outcomes in 7 children. Eur J Pediatr Surg; 2006 Oct;16(5):318-22

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  • [Title] Mediastinal germ cell tumors: clinical aspects and outcomes in 7 children.
  • BACKGROUND: Mediastinal germ cell tumors presenting during childhood are extremely rare.
  • This paper reports on the clinical presentations, method(s) of treatment, complications, results and outcomes in a series of children with mediastinal germ cell tumors.
  • METHODS: A retrospective chart review of 7 children treated between 1971 and 2001 for mediastinal germ cell tumor was carried out.
  • Each patient's surgical treatment, peri- and postoperative complications, histological staging and final outcome were analysed.
  • Their sole treatment consisted of complete surgical excision of the tumor and (part of) the thymus using either median sternotomy or left-sided thoracotomy.
  • All four are alive with no evidence of disease, between 2.5 and 29 years after treatment.
  • Malignant tumors were observed in three patients (1 yolk sac tumor, 1 choriocarcinoma and 1 malignant teratoma).
  • Treatment consisted of either biopsy or debulking followed by chemotherapy (and radiotherapy in 1 case).
  • The patient with yolk sac tumor survived; he is now in remission, 4 years after diagnosis.
  • CONCLUSIONS: Both this study and the literature review testify to the extreme rarity of mediastinal germ cell tumors in childhood.
  • Children with this type of tumor usually are severely symptomatic.
  • Histologically malignant tumors, on the other hand, have a worse prognosis.
  • However, the use of platinum-based combination chemotherapy has considerably increased the survival rates.
  • [MeSH-major] Mediastinal Neoplasms / diagnosis. Mediastinal Neoplasms / surgery. Neoplasms, Germ Cell and Embryonal / diagnosis. Neoplasms, Germ Cell and Embryonal / surgery
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Female. Humans. Infant. Male. Prognosis. Retrospective Studies. Teratoma / diagnosis. Teratoma / surgery. Treatment Outcome

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  • (PMID = 17160775.001).
  • [ISSN] 0939-7248
  • [Journal-full-title] European journal of pediatric surgery : official journal of Austrian Association of Pediatric Surgery ... [et al] = Zeitschrift für Kinderchirurgie
  • [ISO-abbreviation] Eur J Pediatr Surg
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Germany
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6. Arora M, Shrivastav RK, Jaiprakash MP: A rare germ-cell tumor site: vaginal endodermal sinus tumor. Pediatr Surg Int; 2002 Sep;18(5-6):521-3
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  • [Title] A rare germ-cell tumor site: vaginal endodermal sinus tumor.
  • Malignant germ-cell tumors (MGCT) are rare tumors of childhood accounting for less than 3% of pediatric malignancies.
  • Endodermal sinus tumor (EST) forms the most common histologic subtype of MGCT.
  • The serum alpha-fetoprotein (AFP) was elevated partial vaginectomy was done and the tumor was excised in toto.
  • EST of the vagina is a rare, highly malignant GCT that exclusively involves children less than 3 years of age.
  • Partial vaginectomy with combination chemotherapy is the most recommended line of treatment.
  • The surgery eradicates local tumor cells and makes subsequent chemotherapy more effective.
  • Simple tumor excision is not sufficient, as residual tumor cells induce early recurrence and make chemotherapy ineffective.
  • [MeSH-major] Endodermal Sinus Tumor / surgery. Vaginal Neoplasms / surgery
  • [MeSH-minor] Female. Humans. Infant. Tomography, X-Ray Computed

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  • (PMID = 12415399.001).
  • [ISSN] 0179-0358
  • [Journal-full-title] Pediatric surgery international
  • [ISO-abbreviation] Pediatr. Surg. Int.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] Germany
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7. Göbel U, Calaminus G, Schneider DT, Schmidt P, Haas RJ, MAKEI and MAHO Study Groups of the German Society of Pediatric Oncology and Hematology, and the SIOP CNS GCT Study Group: Management of germ cell tumors in children: approaches to cure. Onkologie; 2002 Feb;25(1):14-22
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  • [Title] Management of germ cell tumors in children: approaches to cure.
  • The introduction of cisplatinum chemotherapy and current advances in the surgical treatment have resulted in a dramatic improvement of the prognosis of children with malignant germ cell tumors (GCT).
  • Cisplatinum chemotherapy generally results in sufficient systemic tumor control, but local relapses may still occur in patients who did not receive adequate local treatment.
  • Therefore, the therapeutic consideration must take into account age, primary site of the tumor, and its histology.
  • In gonadal tumors, there is a high chance of primary complete resection since these tumors tend to be encapsulated, and particularly testicular GCT are often detected at a low tumor stage.
  • In these tumors, a neoadjuvant or preoperative chemotherapy after clinical diagnosis by imaging and evaluation of tumor markers significantly facilitates complete resection on delayed surgery.
  • In addition, the impact of chemotherapy on local tumor control may be enhanced by locoregional hyperthermia.
  • In intracranial GCT, radiotherapy significantly contributes to local tumor control, and doses are stratified according to histology.
  • These general considerations have been integrated into national and international cooperative treatment protocols.
  • In most current protocols, treatment is stratified according to an initial risk assessment that includes the parameters age, site, histology, stage, completeness of resection and the tumor markers alpha(1)-fetoprotein (AFP) and human choriogonadotropin (beta-HCG).
  • Moreover, the previously high-risk groups may now expect a favorable prognosis with this risk-adapted treatment, whereas an increasing number of low-risk patients are treated expectantly or with significantly reduced chemotherapy.
  • As current biologic studies reveal distinct genetic patterns in childhood GCT, it can be expected that further combined clinical and genetic studies will be valuable for risk assessment of childhood GCT.
  • [MeSH-major] Neoplasms, Germ Cell and Embryonal / therapy
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Chorionic Gonadotropin, beta Subunit, Human / blood. Cisplatin / administration & dosage. Cisplatin / adverse effects. Combined Modality Therapy. Female. Humans. Infant. Infant, Newborn. Male. Neoadjuvant Therapy. Neoplasm Staging. Prognosis. alpha-Fetoproteins / metabolism

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  • [Copyright] Copyright 2002 S. Karger GmbH, Freiburg
  • (PMID = 11893878.001).
  • [ISSN] 0378-584X
  • [Journal-full-title] Onkologie
  • [ISO-abbreviation] Onkologie
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] Switzerland
  • [Chemical-registry-number] 0 / Chorionic Gonadotropin, beta Subunit, Human; 0 / alpha-Fetoproteins; Q20Q21Q62J / Cisplatin
  • [Number-of-references] 66
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8. Göbel U, Schneider DT, Calaminus G, Haas RJ, Schmidt P, Harms D: Germ-cell tumors in childhood and adolescence. GPOH MAKEI and the MAHO study groups. Ann Oncol; 2000 Mar;11(3):263-71

  • [Source] The source of this record is MEDLINE®, a database of the U.S. National Library of Medicine.
  • [Title] Germ-cell tumors in childhood and adolescence. GPOH MAKEI and the MAHO study groups.
  • In mature and immature teratoma the treatment is surgical.
  • In case of a microscopically complete tumor resection there is no role for adjuvant chemo- or radiotherapy irrespective of the histological grade of immaturity.
  • Malignant germ-cell tumors (GCT) account for 2.9% of all malignant tumors of children younger than 15 years of age.
  • In patients with extensive tumor growth, metastatic disease or secreting intracranial tumors a delayed tumor resection after preoperative chemotherapy is preferable.
  • In these patients malignant non-seminomatous GCT may be diagnosed clinically due to the increased serum or cerebrospinal fluid levels of the tumor markers AFP and/or beta-HCG.
  • Current risk adapted treatment protocols containing cisplatinum allow long-term remissions in about 80% including patients with bulky or metastatic tumors.
  • In the cisplatinum era the prognostic factors like histology, primary site of the tumor and initial tumor stage have partly lost their former impressive significance in infants and children.
  • On the other hand the completeness of the primary tumor resection according to oncological standards has been established as the most powerful prognostic parameter superior to tumor marker levels or primary site of the tumor.

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  • (PMID = 10811491.001).
  • [ISSN] 0923-7534
  • [Journal-full-title] Annals of oncology : official journal of the European Society for Medical Oncology
  • [ISO-abbreviation] Ann. Oncol.
  • [Language] ENG
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Biomarkers; 0 / Biomarkers, Tumor
  • [Number-of-references] 44
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9. Suita S, Shono K, Tajiri T, Takamatsu T, Mizote H, Nagasaki A, Inomata Y, Hara T, Okamura J, Miyazaki S, Kawakami K, Eguchi H, Tsuneyoshi M, Committee for Pediatric Solid Malignant Tumors in the Kyushu Area: Malignant germ cell tumors: clinical characteristics, treatment, and outcome. A report from the study group for Pediatric Solid Malignant Tumors in the Kyushu Area, Japan. J Pediatr Surg; 2002 Dec;37(12):1703-6
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  • [Title] Malignant germ cell tumors: clinical characteristics, treatment, and outcome. A report from the study group for Pediatric Solid Malignant Tumors in the Kyushu Area, Japan.
  • PURPOSE: This study aims to assess the prognostic factors and optimal treatments for malignant germ cell tumors (MGCT) in childhood.
  • The prognostic factors and treatments were assessed based on the 5-year survival rate. RESULTS:.
  • (1) Stage: 100% for stage I (n = 54), 75.0% for stage II (n = 4), 67.3% for stage III (n = 14), and 54.8% for stage IV (n = 33); Unknown: n = 12. (2) Primary site: 93.4% for the testis (n = 52), 86.7% for the ovary (n = 31), 56.9% for the sacrococcygeal (n = 21), and 60.6% for others (n = 12); unknown: n = 1. (3) Surgical intervention for primary tumor: 100% for stage I with a complete resection (n = 53), 78.4% for stage III, IV with a complete resection (n = 26), and 33.3% for stage III, IV with an incomplete resection (n = 21). (4) Type of chemotherapy for the stage III and IV: 83.9% for the PVB (cisplatin, vinblastin, bleomycin; n = 13), 66.7% for the VAC (vincristine, actinomycin D, cyclophosphamide; n = 6), and 47.1% for other regimens (n = 25).
  • Radical complete resection alone is a sufficient treatment for localized MGCT.
  • The PVB regimen is optimal chemotherapy for advanced MGCT; however, high-risk cases still may require more aggressive treatment.
  • [MeSH-major] Germinoma / diagnosis. Germinoma / therapy
  • [MeSH-minor] Adolescent. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Child. Child, Preschool. Female. Humans. Incidence. Infant. Infant, Newborn. Liver Neoplasms / secondary. Lung Neoplasms / secondary. Male. Neoplasm Staging. Ovarian Neoplasms / diagnosis. Ovarian Neoplasms / epidemiology. Ovarian Neoplasms / surgery. Prognosis. Retrospective Studies. Survival Rate. Testicular Neoplasms / diagnosis. Testicular Neoplasms / epidemiology. Testicular Neoplasms / surgery. Treatment Outcome

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  • [Copyright] Copyright 2002, Elsevier Science (USA). All rights reserved.
  • [CommentIn] J Urol. 2003 Sep;170(3):1040 [12926414.001]
  • (PMID = 12483635.001).
  • [ISSN] 1531-5037
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] United States
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10. Guérin S, Hawkins M, Shamsaldin A, Guibout C, Diallo I, Oberlin O, Brugières L, de Vathaire F: Treatment-adjusted predisposition to second malignant neoplasms after a solid cancer in childhood: a case-control study. J Clin Oncol; 2007 Jul 1;25(19):2833-9
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  • [Title] Treatment-adjusted predisposition to second malignant neoplasms after a solid cancer in childhood: a case-control study.
  • PURPOSE: Previous therapy, genetic susceptibility, and the type of first malignant neoplasm (FMN) are known to be associated with the risk of second malignant neoplasm (SMN) among patients treated for a childhood cancer.
  • PATIENTS AND METHODS: A case-control study nested in a European cohort of 4,581 patients treated for a solid cancer during childhood was conducted.
  • One hundred forty-six patients with an SMN and 417 controls were matched for sex, age at FMN, chemotherapy, radiotherapy, the local radiation dose received at the site of SMN for patient cases and at the same site for the matched controls, and follow-up.
  • RESULTS: A significantly increased risk of developing any SMN was observed after Hodgkin's lymphoma, retinoblastoma, malignant bone tumor, soft tissue sarcoma (STS), and germ cell tumor as FMN, after adjustment for chemotherapy and family cancer syndrome.
  • No significant risk of developing a carcinoma was observed among patients who had developed Hodgkin's lymphoma as FMN.
  • A significantly increased risk of developing a sarcoma was observed among patients who had developed a retinoblastoma (adjusted odds ratio [ORa] = 7.5; 95% CI, 1.2 to 46), a malignant bone tumor (ORa = 13.3; 95% CI, 1.5 to 117), an STS (ORa = 4.8; 95% CI, 1.3 to 18), or a carcinoma (ORa = 9.4; 95% CI, 1.1 to 82) as FMN.
  • CONCLUSION: Survivors of Hodgkin's lymphoma, retinoblastoma, malignant bone tumor, STS, and germ cell tumor should receive close surveillance because they are at increased risk of developing any SMN.
  • [MeSH-major] Hodgkin Disease / therapy. Neoplasms / drug therapy. Neoplasms / radiotherapy. Neoplasms, Germ Cell and Embryonal / therapy. Neoplasms, Second Primary / etiology. Retinoblastoma / therapy. Sarcoma / therapy


11. De Backer A, Madern GC, Oosterhuis JW, Hakvoort-Cammel FG, Hazebroek FW: Ovarian germ cell tumors in children: a clinical study of 66 patients. Pediatr Blood Cancer; 2006 Apr;46(4):459-64
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  • [Title] Ovarian germ cell tumors in children: a clinical study of 66 patients.
  • BACKGROUND: Ovarian germ cell tumors are rare in childhood.
  • The aim of this study is to review clinical presentation, management, and outcome in a two-center series of girls with ovarian germ cell tumor.
  • PROCEDURE: The records of 66 patients (median age 9 years) with histologically proven ovarian germ cell tumor (either benign or malignant), treated over a 44-year-span, were reviewed.
  • Sixteen patients had an emergency operation for tumor torsion.
  • Unilateral salpingo-oophorectomy was the most frequently performed procedure (n = 46), and ovarian-sparing tumorectomy was performed in 9 patients (one bilaterally).
  • Surgical removal of the tumor with or without the ovary and/or adnex was the sole treatment in 55 patients, chemotherapy was administered in 10 and radiotherapy + chemotherapy in one.
  • Two patients, with malignant disease, died.
  • The 64 survivors are now between 8 months and 44 years after treatment.
  • CONCLUSIONS: With a recurrence rate of 4.5% and a mortality rate of 3%, this series confirms the excellent prognosis for girls with ovarian germ cell tumor (GCT).
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Neoplasms, Germ Cell and Embryonal / drug therapy. Ovarian Neoplasms / drug therapy
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Female. Humans. Infant. Infant, Newborn. Magnetic Resonance Imaging. Neoplasm Staging. Tomography, X-Ray Computed. Treatment Outcome

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  • (PMID = 16206211.001).
  • [ISSN] 1545-5009
  • [Journal-full-title] Pediatric blood & cancer
  • [ISO-abbreviation] Pediatr Blood Cancer
  • [Language] eng
  • [Publication-type] Journal Article; Multicenter Study
  • [Publication-country] United States
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12. Shen K, Lang J, Huang H: [Treatment of childhood genital malignancies]. Zhonghua Fu Chan Ke Za Zhi; 2001 Jun;36(6):360-3
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  • [Title] [Treatment of childhood genital malignancies].
  • OBJECTIVE: To present the clinical profile and the result of treatment in childhood genital malignancies.
  • METHODS: Twenty seven childhood patients (< 12 years) with genital malignancies diagnosed and treated in Peking Union Medical College hospital were analyzed retrospectively focusing on the clinical characteristics and outcome of the treatment.
  • Of the 27 patients, 23 had ovarian malignancies including 21 (77.8%) germ cell tumor.
  • Four of the patients had extragonadal tumors with 2 endodermal sinus tumor of the vagina, and 2 embryonic rhabdomysarcoma.
  • In vaginal malignancies, vaginal bleeding and polypoid tumor of the vagina were important clinical manifestations.
  • Childhood genital malignancies were very sensitive to the chemotherapy.
  • Postoperative vincristine, actinomycine, cyclophosphamide (VAC) or cisplatine, vincristine, blyomycine (PVB) chemotherapy was individualized according to the tumor site, International Federation of Gynecology and obstetrics stage and histopathology.
  • Survival rate in our series was 71.4% during the follow-up time (mean 47.4 months).
  • CONCLUSIONS: Malignant germ cell tumor of the ovary is the most common tumor in the childhood genital malignancies.
  • Principle of primary surgery is preservation of fertility and chemotherapy is also considered as a very important treatment modality.
  • [MeSH-major] Genital Neoplasms, Female / therapy
  • [MeSH-minor] Antineoplastic Combined Chemotherapy Protocols / administration & dosage. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Bleomycin / administration & dosage. Child. Child, Preschool. Cisplatin / administration & dosage. Combined Modality Therapy. Cyclophosphamide / administration & dosage. Dactinomycin / administration & dosage. Drug Administration Schedule. Female. Humans. Retrospective Studies. Vinblastine / administration & dosage. Vincristine / administration & dosage

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  • (PMID = 11783136.001).
  • [ISSN] 0529-567X
  • [Journal-full-title] Zhonghua fu chan ke za zhi
  • [ISO-abbreviation] Zhonghua Fu Chan Ke Za Zhi
  • [Language] chi
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] China
  • [Chemical-registry-number] 11056-06-7 / Bleomycin; 1CC1JFE158 / Dactinomycin; 5J49Q6B70F / Vincristine; 5V9KLZ54CY / Vinblastine; 8N3DW7272P / Cyclophosphamide; Q20Q21Q62J / Cisplatin; PVB protocol; VAC protocol
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13. Tang JY, Pan C, Xu M: [Protocol for diagnosis and treatment of childhood malignant germ cell tumor]. Zhonghua Er Ke Za Zhi; 2008 May;46(5):388-9
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  • [Title] [Protocol for diagnosis and treatment of childhood malignant germ cell tumor].
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Neoplasms, Germ Cell and Embryonal / diagnosis. Neoplasms, Germ Cell and Embryonal / drug therapy
  • [MeSH-minor] Child. Child, Preschool. Female. Humans. Infant. Male. Ovarian Neoplasms / diagnosis. Ovarian Neoplasms / drug therapy. Testicular Neoplasms / diagnosis. Testicular Neoplasms / drug therapy

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  • (PMID = 19099762.001).
  • [ISSN] 0578-1310
  • [Journal-full-title] Zhonghua er ke za zhi = Chinese journal of pediatrics
  • [ISO-abbreviation] Zhonghua Er Ke Za Zhi
  • [Language] chi
  • [Publication-type] Journal Article
  • [Publication-country] China
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14. Göbel U, Calaminus G, Schneider DT, Koch S, Teske C, Harms D: The malignant potential of teratomas in infancy and childhood: the MAKEI experiences in non-testicular teratoma and implications for a new protocol. Klin Padiatr; 2006 Nov-Dec;218(6):309-14
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  • [Title] The malignant potential of teratomas in infancy and childhood: the MAKEI experiences in non-testicular teratoma and implications for a new protocol.
  • Since 1982, mature and immature teratomas have been recruited into the MAHO and MAKEI protocols of the German Society for Pediatric Oncology and Hematology (GPOH) for testicular and non-testicular germ cell tumors in order to study the epidemiology and clinical behaviour of teratomas.
  • Patients were registered in the epidemiologic German Childrens Cancer Registry and the GPOH Childrens Tumor Registry for pathological review.
  • Patients with immaturity grade 2 and 3 according to Gonzales-Crussi were eligible for adjuvant chemotherapy.
  • 1) EFS after complete tumor resection has been estimated to 0.96 +/- 0.01 in both observation periods.
  • 2) Incomplete tumor resection remains the main risk factor for relapse (EFS 0.55 +/- 0.09).
  • 4) In MAKEI 83/86/89 four newborns with teratoma died due to perioperative complications and nine children as a result of tumor progression, whereas in MAKEI 96 no newborn died, only one child died from tumor progression, and another child died during long time observation for another reason (meningitis).
  • 5) In accordance to the experience of the MAKEI 83/86/89 studies, no child of the MAKEI 96 study presented with yolk sac tumor at recurrence if adjuvant chemotherapy was administered during first-line treatment because of immaturity.
  • In contrast, more than half of the children with tumor recurrence after watch and wait strategy had yolk sac tumor in addition to teratoma.
  • [MeSH-minor] Adolescent. Adult. Age Factors. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Chemotherapy, Adjuvant. Chi-Square Distribution. Child. Child, Preschool. Data Interpretation, Statistical. Disease Progression. Female. Humans. Infant. Infant, Newborn. Male. Neoplasm Recurrence, Local. Neoplasm Staging. Neoplasms, Germ Cell and Embryonal / pathology. Ovary / pathology. Prospective Studies. Randomized Controlled Trials as Topic. Sacrococcygeal Region. Sex Factors. Treatment Outcome. World Health Organization

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  • (PMID = 17080332.001).
  • [ISSN] 0300-8630
  • [Journal-full-title] Klinische Pädiatrie
  • [ISO-abbreviation] Klin Padiatr
  • [Language] eng
  • [Publication-type] Comparative Study; Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Germany
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15. Cai JY, Tang JY, Pan C, Xu M, Xue HL, Zhou M, Dong L, Ye QD, Jiang H, Shen SH, Chen J: Results of RS-99 protocol for childhood solid tumors. World J Pediatr; 2010 Feb;6(1):43-9
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  • [Title] Results of RS-99 protocol for childhood solid tumors.
  • BACKGROUND: Little was known about the therapeutic result of rhabdomyosarcomas (RMSs) and other malignant tumors until the end of the last century in China.
  • We designed a RS-99 protocol under close cooperation of a multidisciplinary team including surgeons, radiologists, pathologists, and pediatric oncologists at Shanghai Children's Medical Center.
  • This study aimed to improve the prognosis of childhood solid tumors and analyze the results of different tumors with the same protocol, including RMSs, the Ewing sarcoma family of tumors (ESFTs), and ex-cranial germ cell tumors (GCTs).
  • METHODS: Sixty-six patients with malignant solid tumors [RMS (n=30), GCT (n=22), and ESFT (n=14)] were enrolled on the RS-99 protocol from October 1998 to October 2006.
  • The protocol involved surgery, radiotherapy and chemotherapy which included VCP (vincristine, cisdiaminedichloroplatinum, and cyclophosphamide) and IEV (etoposide, vincristine and ifosfamide) for the low-risk group, AVCP (adriamycin, vincristine, cisdiaminedichloroplatinum, and cyclophosphamide) and IEV for the intermediate-risk group and high-risk group.
  • Peripheral blood stem cell transplantation was suggested for the high-risk group.
  • Tumor stage and risk group were also contributive to prognosis (P=0.008).
  • For GCT patients, the primary sites of tumors and their histological classification did not influence the therapeutic result (P=0.814).
  • One ESFT patient developed a second cancer.
  • [MeSH-major] Bone Neoplasms / therapy. Neoplasms, Germ Cell and Embryonal / therapy. Rhabdomyosarcoma / therapy. Sarcoma, Ewing / therapy
  • [MeSH-minor] Adolescent. Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Chemotherapy, Adjuvant. Child. Child, Preschool. Cyclophosphamide / therapeutic use. Disease-Free Survival. Doxorubicin / therapeutic use. Epirubicin / therapeutic use. Etoposide / therapeutic use. Female. Humans. Ifosfamide / therapeutic use. Infant. Male. Peripheral Blood Stem Cell Transplantation. Prednisolone / therapeutic use. Prognosis. Radiotherapy, Adjuvant. Tamoxifen / therapeutic use. Vincristine / therapeutic use

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  • (PMID = 20143210.001).
  • [ISSN] 1867-0687
  • [Journal-full-title] World journal of pediatrics : WJP
  • [ISO-abbreviation] World J Pediatr
  • [Language] eng
  • [Publication-type] Clinical Trial; Journal Article
  • [Publication-country] Switzerland
  • [Chemical-registry-number] 094ZI81Y45 / Tamoxifen; 3Z8479ZZ5X / Epirubicin; 5J49Q6B70F / Vincristine; 6PLQ3CP4P3 / Etoposide; 80168379AG / Doxorubicin; 8N3DW7272P / Cyclophosphamide; 9PHQ9Y1OLM / Prednisolone; UM20QQM95Y / Ifosfamide; AVCT protocol; IEV protocol; VCP protocol 2
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16. Kumar V, Kini P, Vepakomma D, Basant M: Vaginal endodermal sinus tumor. Indian J Pediatr; 2005 Sep;72(9):797-8
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  • [Title] 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).
  • Magnetic Resonance Imaging (MRI) showed a tumor mass at the junction of cervix and vagina and biopsy was suggestive of a vaginal EST.
  • The tumor was excised in toto and patient received four courses of cisplatin, etoposide and bleomycin.
  • EST of the vagina is a rare, highly malignant GCT that exclusively involves children less than 3 years of age.
  • 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.
  • [MeSH-major] Endodermal Sinus Tumor / diagnosis. Vaginal Neoplasms / diagnosis

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  • (PMID = 16186686.001).
  • [ISSN] 0973-7693
  • [Journal-full-title] Indian journal of pediatrics
  • [ISO-abbreviation] Indian J Pediatr
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] India
  • [Chemical-registry-number] 0 / alpha-Fetoproteins
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17. Rzepka-Górska I, Błogowska A, Zajaczek S, Zielińska D: [Germinal cell tumors in young and adolescent girls]. Ginekol Pol; 2003 Sep;74(9):840-6
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  • [Title] [Germinal cell tumors in young and adolescent girls].
  • OBJECTIVE: Germ cell tumours are the most common ovarian tumours in childhood and adolescence.
  • This diverse group of tumours derives from germ cells.
  • DESIGN: The aim of this work is presentation of germ cell tumours in the material from our clinic with characteristic clinical features, the scope of operation and effects of many years of observation.
  • MATERIALS AND METHODS: We treated 109 girls with germ cell tumours of the ovary: 13 had malignant tumours: there were 7 patients with dysgerminomas, 2 with endodermal sinus tumour of the ovary, 3 with immature teratomas, 1 with carcinoma embryonale.
  • It must be suggested that patients of stage I who wish to preserve childbearing function may be treated with unilateral salpingoophorectomy and adjuvant chemotherapy.
  • Monitoring of the treatment is connected with measurement of biochemical markers.
  • Some of these markers are useful for monitoring of response to therapy.
  • CONCLUSIONS: The sift ultrasonographic investigations can be helpful in the early diagnosis of germ cell tumours of the ovary in girls.
  • Fertility sparing operative treatment is preferred when karyotype is normal.
  • [MeSH-major] Neoplasms, Germ Cell and Embryonal / pathology. Neoplasms, Germ Cell and Embryonal / therapy. Ovarian Neoplasms / pathology. Ovarian Neoplasms / therapy
  • [MeSH-minor] Adolescent. Adult. Biomarkers, Tumor / blood. Child. Combined Modality Therapy. Dysgerminoma / pathology. Dysgerminoma / therapy. Endodermal Sinus Tumor / pathology. Endodermal Sinus Tumor / therapy. Female. Germinoma / pathology. Germinoma / therapy. Gonadoblastoma / pathology. Gonadoblastoma / therapy. Gonadotropins / blood. Gonadotropins / genetics. Humans. Poland. Risk Factors. Teratoma / pathology. Teratoma / therapy

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  • (PMID = 14674134.001).
  • [ISSN] 0017-0011
  • [Journal-full-title] Ginekologia polska
  • [ISO-abbreviation] Ginekol. Pol.
  • [Language] pol
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Poland
  • [Chemical-registry-number] 0 / Biomarkers, Tumor; 0 / Gonadotropins
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18. Tansel T, Onursal E, Dayloğlu E, Başaran M, Sungur Z, Qamci E, Yilmazbayhan D, Eker R, Ertuğrul T: Childhood mediastinal masses in infants and children. Turk J Pediatr; 2006 Jan-Mar;48(1):8-12

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  • [Title] Childhood mediastinal masses in infants and children.
  • We reviewed all cases of primary pediatric mediastinal masses diagnosed and treated over a 24-year period.
  • In this study, out of 187 primary mediastinal mass cases diagnosed between 1980 and 2004 in Istanbul University Istanbul Faculty of Medicine, Cardiovascular Surgery Department, 37 pediatric primary mediastinal mass cases were retrospectively evaluated according to age, sex, symptoms, diagnostic procedure, anatomical location, surgical treatment, histopathological evaluation and postoperative adjuvant therapy.
  • The patients ranged in age from 2 months to 15 years at the time of diagnosis, with a mean age of 8 years.
  • There were 24 benign (64.8%) and 13 malignant (35.2%) tumors.
  • The cases were lymphoma (27%), neurogenic tumors (21.6%), cystic lesions (18.9%), germ cell tumors (13.5%), thymic lesions (10.8%) and cardiac tumors (8.1%).
  • Complete and partial resections of the tumor were the surgical procedures performed in 24 patients (64.8%) and 3 patients (8.1%), respectively.
  • The three patients with a malignant tumor, in whom the entire mass could not be removed, received chemotherapy and radiation after surgery.
  • In 10 patients with lymphoma, surgery was not a part of treatment and they received medical and radiation therapy after the establishment of the definitive diagnosis.
  • Primary pediatric mediastinal malignancies are relatively common in infants and children.

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  • (PMID = 16562779.001).
  • [ISSN] 0041-4301
  • [Journal-full-title] The Turkish journal of pediatrics
  • [ISO-abbreviation] Turk. J. Pediatr.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] Turkey
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19. Schneider DT, Calaminus G, Göbel U: Diagnostic value of alpha 1-fetoprotein and beta-human chorionic gonadotropin in infancy and childhood. Pediatr Hematol Oncol; 2001 Jan-Feb;18(1):11-26
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  • [Title] Diagnostic value of alpha 1-fetoprotein and beta-human chorionic gonadotropin in infancy and childhood.
  • This article provides a comprehensive review of the current literature and summarizes the experience of the German cooperative protocols for nontesticular germ cell tumors (MAKEI) on the use of alpha 1-fetoprotein (AFP) and beta-human chorionic gonadotropin (beta-hCG) for diagnostic evaluation in pediatric oncology.
  • AFP and beta-hCG represent the characteristic tumor markers of malignant epithelial liver tumors and malignant germ cell tumors (GCT).
  • They play an important role in the initial diagnostic evaluation as well as in the follow-up examination during therapy.
  • Current therapeutic strategies for both tumor types include preoperative chemotherapy followed by delayed tumor resection.
  • In a well-defined clinical setting, these tumor markers allow clinical diagnosis without histological confirmation, but the physiologically elevated AFP serum levels in infancy or unrecognized benign conditions, such as hepatic diseases or hereditary disorders, must also be considered as differential diagnoses.
  • Having excluded these conditions, elevated AFP levels above the age-related normal range (with or without high beta-hCG), indicate malignant epithelial liver tumors in primary liver lesions.
  • At virtually all other sites, the diagnosis of a malignant GCT with a substantial yolk sac tumor and/or choriocarcinoma component can be established.
  • Other childhood tumors are rarely associated with AFP or beta-hCG production, usually at only moderately elevated serum levels, and in most patients these tumors can be excluded by clinical and radiological examination.
  • [MeSH-major] Biomarkers, Tumor / analysis. Chorionic Gonadotropin, beta Subunit, Human / analysis. Neoplasms / diagnosis. alpha-Fetoproteins / analysis
  • [MeSH-minor] Child. Endodermal Sinus Tumor / diagnosis. Humans. Infant. Infant, Newborn. Liver Neoplasms / diagnosis

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  • (PMID = 11205836.001).
  • [ISSN] 0888-0018
  • [Journal-full-title] Pediatric hematology and oncology
  • [ISO-abbreviation] Pediatr Hematol Oncol
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] England
  • [Chemical-registry-number] 0 / Biomarkers, Tumor; 0 / Chorionic Gonadotropin, beta Subunit, Human; 0 / alpha-Fetoproteins
  • [Number-of-references] 94
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20. Szavay PO, Wermes C, Fuchs J, Schrappe M, Flemming P, von Schweinitz D: Effective treatment of infantile choriocarcinoma in the liver with chemotherapy and surgical resection: a case report. J Pediatr Surg; 2000 Jul;35(7):1134-5
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  • [Title] Effective treatment of infantile choriocarcinoma in the liver with chemotherapy and surgical resection: a case report.
  • To the authors' knowledge, this is the first report on successful treatment with preoperative chemotherapy.
  • A 10-week-old girl presented with a large liver tumor, ovarian cysts, cardiac insufficiency, progressive hemolytic anemia, and thrombocytopenia.
  • Ultrasound scan and magnetic resonance tomography (MRT) showed the typical pattern of infantile hemangioendothelioma.
  • An emergency laparotomy was performed because of increasing cardiac insufficiency with ligation of the right hepatic artery, tumor biopsy, and subtotal resection of the ovarian cysts.
  • Chemotherapy was initiated with etoposide and cisplatin.
  • When x-ray examination showed development of lung metastases, chemotherapy was intensified with etoposide, cisplatin, and ifosfamid according to the German Study Group of Extracranial Nontesticular Malignant Germ Cell Tumors in Childhood and Adolescence (MAKEI-96).
  • Serum beta-HCG levels decreased further, ultrasound examination showed significant tumor reduction, and pulmonal metastasis could no longer be found in chest x-rays.
  • After the fourth course, a complete tumor resection was achieved by an extended right hemihepatectomy with adjuvant chemotherapy being administered after the operation.
  • The authors' experience shows that chemotherapy is effective for preoperative tumor reduction.
  • [MeSH-major] Choriocarcinoma / drug therapy. Choriocarcinoma / surgery. Liver Neoplasms / drug therapy. Liver Neoplasms / surgery
  • [MeSH-minor] Combined Modality Therapy. Female. Humans. Infant, Newborn. Remission Induction

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  • (PMID = 10917316.001).
  • [ISSN] 0022-3468
  • [Journal-full-title] Journal of pediatric surgery
  • [ISO-abbreviation] J. Pediatr. Surg.
  • [Language] eng
  • [Publication-type] Case Reports; Journal Article
  • [Publication-country] UNITED STATES
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21. Schneider DT, Calaminus G, Wessalowski R, Pathmanathan R, Harms D, Göbel U: Therapy of advanced ovarian juvenile granulosa cell tumors. Klin Padiatr; 2002 Jul-Aug;214(4):173-8
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  • [Title] Therapy of advanced ovarian juvenile granulosa cell tumors.
  • BACKGROUND: Gonadal sex cord-stromal tumors are rare tumors that develop from the gonadal non-germ cell component such as granulosa, Sertoli or Leydig cells.
  • Among these, juvenile granulosa cell tumors (JGCT) constitute the largest subgroup of ovarian sex cord-stromal tumors during childhood and adolescence.
  • In local disease (FIGO stage I), the beneficial role of tumor-ovarectomy is well established.
  • In contrast, life expectancy in patients with advanced JGCT (FIGO stage >/= II) is short even after complete tumor resection.
  • The current literature provides only limited and inconclusive data regarding the value of adjuvant chemotherapy in such patients with advanced disease.
  • PATIENTS AND METHODS: Therefore, we analyzed the patients with FIGO stage >/= II JGCT who were prospectively documented as follow-up patients of the German MAKEI trials for non-testicular germ cell tumors and received the recommended cisplatin-based chemotherapy in an adjuvant setting.
  • Two patients received laparoscopic tumor resection, which was incomplete in both.
  • All patients received 4 or 6 cycles of adjuvant cisplatin-based three-agent chemotherapy in analogy to the current therapeutic concept applied in malignant germ cell tumors.
  • One patient with a large tumor and multiple peritoneal metastases additionally received 40 Gy abdominal irradiation.
  • RESULTS: All patients achieved complete clinical remission after initial surgery and adjuvant chemotherapy.
  • One patient developed a metachronous tumor of the contralateral ovary after 126 months follow-up and is still alive but currently in therapy of another recurrence.
  • Another patient suffered a tumor recurrence after 12 months but achieved a second complete remission with cisplatin chemotherapy after a follow-up of currently 4 months.
  • One patient achieved complete clinical remission but suffered a diffuse peritoneal tumor recurrence with massive ascites and finally died as a result of tumor progression.
  • In summary, at the time of this report 6 of 7 patients are alive after a median of 47 (15 - 138) months.
  • CONCLUSION: This analysis clearly demonstrates that advanced JGCT can be successfully treated with surgery followed by adjuvant cisplatin-based chemotherapy.
  • Therefore, this study reveals encouraging therapeutic perspectives in these otherwise fatal tumors that merit further investigation in a prospective cooperative trial.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Granulosa Cell Tumor / drug therapy. Ovarian Neoplasms / drug therapy
  • [MeSH-minor] Adolescent. Chemotherapy, Adjuvant. Child. Cisplatin / administration & dosage. Cisplatin / adverse effects. Combined Modality Therapy. Female. Follow-Up Studies. Germany. Humans. Neoplasm Staging. Prospective Studies. Survival Rate

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  • (PMID = 12165898.001).
  • [ISSN] 0300-8630
  • [Journal-full-title] Klinische Pädiatrie
  • [ISO-abbreviation] Klin Padiatr
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't
  • [Publication-country] Germany
  • [Chemical-registry-number] Q20Q21Q62J / Cisplatin
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22. Cheng HY, Zhu XD, Wang HM, Qin H: [Application of ATP based bioluminescence tumor chemosensitivity assay in the chemotherapy of pediatric solid tumor]. Zhonghua Er Ke Za Zhi; 2009 Aug;47(8):598-603
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  • [Title] [Application of ATP based bioluminescence tumor chemosensitivity assay in the chemotherapy of pediatric solid tumor].
  • OBJECTIVE: To explore the clinical significance of ATP based bioluminescence in vitro tumor chemosensitivity assay (ATP-TCA) in the chemotherapy of pediatric solid tumor.
  • METHODS: The cell culture technique and ATP-TCA were used to study chemosensitivity assay in specimens from 50 cases who underwent resection surgery for solid tumor (15 malignant neurogenic tumor, 8 malignant germ cell tumors, 10 Wilms' tumors, 10 hepatoblastomas, 6 rhabdomyosarcomas, 1 adrenocortical carcinoma), 8 chemotherapeutic drugs and 8 drug combination schedules were applied in every specimen. RESULTS:.
  • (1) Specimens of 46 of 50 pediatric patients with solid tumors were suitable for evaluation and were evaluated, the overall evaluation rate was 92% (46/50). (2) There was the heterogeneity in the chemosensitivity of the solid tumors in vitro. (3) The drug combination schedules of high sensitivity rate of every kind of pediatric solid tumor are as follows: the malignant neurogenic tumor: CBP + EPI + IFO (12/15, 80.0%), VCR + CTX + DDP + DTIC (11/15, 73.3%); malignant germ cell tumor: DDP + VCR + BLM(8/8, 100%), TPTN + ACTD + IFO(8/8, 100%), As2O3 (7/8, 87.5%); Wilms' tumor: VCR + ACTD(6/7, 85.7%), CBP + VP16 (6/8, 75.0%); hepatoblastoma: VCR + CTX + DDP + VP16 (8/9, 88.9%), CBP +IFO + VM26 (7/9, 77.8%), DDP + VP16 + TPTN(7/9, 77.8%); rhabdomyosarcoma: VCR + CTX + DDP + VP16 (5/5, 100%); adrenocortical carcinoma: VCR + CTX + ADM. (4) As2O3 reached a high in vitro sensitive rate of 87.5% (7/8) and 46.7% (7/15) in malignant germ cell tumor and the malignant neurogenic tumor respectively, PTX was sensitive to the malignant neurogenic tumor and rhabdomyosarcoma (40.0% (6/15), 60.0% (3/5)), GEM was sensitive to pediatric malignant germ cell tumor and rhabdomyosarcoma (50.0% (4/8), 60.0% (3/5)).
  • CONCLUSIONS: ATP-TCA is a sensitive method for the chemotherapeutic agents screening of pediatric malignant solid tumor, and ATP-TCA assay results correlated well with clinical response.
  • It appears to be useful in screening new drugs for pediatric solid tumor, exploring the possible combination plots and principles, evaluating the efficacy of existing chemotherapy, and optimize chemotherapy on an individual basis.
  • [MeSH-major] Drug Screening Assays, Antitumor / methods

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  • (PMID = 19951493.001).
  • [ISSN] 0578-1310
  • [Journal-full-title] Zhonghua er ke za zhi = Chinese journal of pediatrics
  • [ISO-abbreviation] Zhonghua Er Ke Za Zhi
  • [Language] chi
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] China
  • [Chemical-registry-number] 0 / Antineoplastic Agents
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23. Handel LN, Scott SM, Giller RH, Greffe BS, Lovell MA, Koyle MA: New perspectives on therapy for vaginal endodermal sinus tumors. J Urol; 2002 Aug;168(2):687-90
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  • [Title] New perspectives on therapy for vaginal endodermal sinus tumors.
  • PURPOSE: Malignant germ cell tumors account for 3% of childhood cancers.
  • Endodermal sinus tumor, the most common malignant germ cell tumor, requires treatment primarily with chemotherapy and surgery is reserved as a last resort.
  • It is rare for the vagina to be the primary site of endodermal sinus tumor, and we report on our experiences with this phenomenon at a single institution.
  • MATERIALS AND METHODS: We retrospectively reviewed the clinical features, treatment and outcomes of 3 children with vaginal endodermal sinus tumor.
  • RESULTS: Initial treatment was combination chemotherapy, in 2 patients. alpha-fetoprotein decreased rapidly and returned to normal in both.
  • One patient is disease-free 7 years after chemotherapy and the other patient is currently disease-free with a normal alpha-fetoprotein 3 months after induction chemotherapy.
  • In the remaining case progressive disease developed following initial chemotherapy and subsequent salvage surgery combined with radiation, chemotherapy and ultimately autologous bone marrow transplant was performed.
  • The patient has remained disease-free for 6.5 years since completing this extensive therapy.
  • CONCLUSIONS: Endodermal sinus tumor of the vagina is rare.
  • Bone marrow transplant should be considered as a last therapeutic resort in salvage cases of unresponsive vaginal endodermal sinus tumor.
  • [MeSH-major] Endodermal Sinus Tumor / therapy. Vaginal Neoplasms / therapy
  • [MeSH-minor] Combined Modality Therapy. Female. Follow-Up Studies. Humans. Infant. Neoadjuvant Therapy. Neoplasm Staging. Retrospective Studies. Salvage Therapy. Survival Rate. Vagina / pathology

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  • (PMID = 12131350.001).
  • [ISSN] 0022-5347
  • [Journal-full-title] The Journal of urology
  • [ISO-abbreviation] J. Urol.
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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24. Ishibashi M, Nakayama K, Oride A, Yeasmin S, Katagiri A, Iida K, Nakayama N, Miyazaki K: [A case of PEP(BEP)-resistant ovarian dysgerminoma successfully treated by VeIP therapy]. Gan To Kagaku Ryoho; 2009 Mar;36(3):513-7
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  • [Title] [A case of PEP(BEP)-resistant ovarian dysgerminoma successfully treated by VeIP therapy].
  • Ovarian germ cell tumors are malignant tumors which commonly develop during childhood, and which are sensitive to chemotherapy.
  • We have had a case of germ cell tumors which showed resistance to first-line PEP(BEP)chemotherapy.
  • As second-line chemotherapy, VeIP therapy was used, because it is possible that this therapy is effective against recurrent testicular germ cell tumors.
  • She experienced acute abdominal pain and visited the hospital, where she was diagnosed with torsion of an ovarian tumor.
  • Then she received chemotherapy PEP(BEP), but after eight months of PEP (BEP), her serum hCG-CTP was again elevated to 14.5 mIU/mL.
  • After the operation, the patient again underwent chemotherapy.
  • After 6 courses of this therapy, she had a follow-up operation.
  • She remains without recurrence of this disease 24 months after VeIP therapy.
  • This case suggests that VeIP therapy might be an effective second-line therapy for patients with PEP(BEP)-resistant ovarian dysgerminoma.
  • [MeSH-major] Antineoplastic Combined Chemotherapy Protocols / therapeutic use. Drug Resistance, Neoplasm / drug effects. Dysgerminoma / drug therapy. Dysgerminoma / pathology. Ovarian Neoplasms / drug therapy. Ovarian Neoplasms / pathology
  • [MeSH-minor] Adolescent. Bleomycin / therapeutic use. Cisplatin / therapeutic use. Female. Humans. Magnetic Resonance Imaging. Positron-Emission Tomography. Remission Induction. Tomography, X-Ray Computed. Treatment Outcome

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  • (PMID = 19295284.001).
  • [ISSN] 0385-0684
  • [Journal-full-title] Gan to kagaku ryoho. Cancer & chemotherapy
  • [ISO-abbreviation] Gan To Kagaku Ryoho
  • [Language] jpn
  • [Publication-type] Case Reports; English Abstract; Journal Article
  • [Publication-country] Japan
  • [Chemical-registry-number] 11056-06-7 / Bleomycin; Q20Q21Q62J / Cisplatin
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25. Shalaby T, Hiyama E, Grotzer MA: Telomere maintenance as therapeutic target in embryonal tumours. Anticancer Agents Med Chem; 2010 Mar;10(3):196-212
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  • [Title] Telomere maintenance as therapeutic target in embryonal tumours.
  • Embryonal tumours most commonly occur in the first few years of life and account for approximately 30% of childhood malignancies.
  • Knowledge of these tumours' genetics has already impacted on their clinical management and further knowledge of their cellular immortalization will hopefully result in novel therapies.
  • A critical length of telomere repeats is required to ensure proper telomere function and avoid the activation of DNA damage pathways that result in senescence and cell death.
  • Hence stabilization of telomere is an important step in cell immortalization and carcinogenesis.
  • Telomere maintenance is evident in virtually all types of malignant cells, including embryonal tumours, where either a telomerase-dependent or alternative lengthening of telomeres (ALT) mechanism is employed in order to ensure their limitless replicative potential.
  • In this review, we are giving an overview about telomere maintenance in childhood tumours and discussing its potential as a new therapeutic target.
  • [MeSH-major] Antineoplastic Agents / therapeutic use. Neoplasms, Germ Cell and Embryonal / drug therapy. Telomerase / physiology. Telomere / metabolism
  • [MeSH-minor] Animals. Child. Embryo, Mammalian / enzymology. G-Quadruplexes / drug effects. Hepatoblastoma / genetics. Humans. Medulloblastoma / genetics. Mice. Neuroblastoma / genetics. Rhabdomyosarcoma / genetics. Sarcoma, Ewing / genetics. Wilms Tumor / genetics

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  • (PMID = 20017721.001).
  • [ISSN] 1875-5992
  • [Journal-full-title] Anti-cancer agents in medicinal chemistry
  • [ISO-abbreviation] Anticancer Agents Med Chem
  • [Language] eng
  • [Publication-type] Journal Article; Research Support, Non-U.S. Gov't; Review
  • [Publication-country] Netherlands
  • [Chemical-registry-number] 0 / Antineoplastic Agents; EC 2.7.7.49 / TERT protein, human; EC 2.7.7.49 / Telomerase; EC 2.7.7.49 / Tert protein, mouse
  • [Number-of-references] 250
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26. Schmidt D, Kommoss F: [Teratoma of the ovary. Clinical and pathological differences between mature and immature teratomas]. Pathologe; 2007 May;28(3):203-8
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  • [Transliterated title] Teratome des Ovars. Klinisch-pathologische Unterschiede zwischen unreifen und reifen Teratomen.
  • Teratomas are the most frequent germ cell tumors of the ovary.
  • Mature teratomas are benign tumors, which are most often composed of derivatives of two or three germ cell layers.
  • Only in rare cases is the transition into a malignant tumor observed (most often squamous cell carcinoma).
  • In contrast, immature teratomas are malignant ovarian tumors.
  • They contain immature tissue elements in addition to the mature components, most often consisting of immature neural tissue.
  • Histologically, this tumor component can be identified as neurotubules or rosettes.
  • The proportion of immature tissue elements defines the grade of immaturity.
  • With the exception of childhood cases, grade 2 and 3 immature teratomas are treated with chemotherapy.
  • In childhood cases, foci of yolk sac tumor (YST) must be looked for, since this determines the prognosis.
  • If a focus of YST is present, the patient is treated with chemotherapy.
  • In cases of immature peritoneal implants, patients are also treated with chemotherapy.

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  • (PMID = 17396268.001).
  • [ISSN] 0172-8113
  • [Journal-full-title] Der Pathologe
  • [ISO-abbreviation] Pathologe
  • [Language] ger
  • [Publication-type] English Abstract; Journal Article
  • [Publication-country] Germany
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27. Islam S, Yamout SZ, Gosche JR: Management and outcomes of ovarian masses in children and adolescents. Am Surg; 2008 Nov;74(11):1062-5
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  • Ovarian masses in the pediatric age group are rare, and malignancies are even less common.
  • Demographic and tumor-specific data were reviewed and analyzed, and a Student's unpaired t test was used where appropriate.
  • Eight masses were malignant (16%) with malignant teratoma, dysgerminoma, and germ cell tumors found.
  • These patients responded to chemotherapy, but there were three recurrences noted that responded to further therapy.
  • A majority of the patients underwent a laparotomy with 12 per cent having a minimally invasive procedure.
  • Only 37 per cent of the operations were performed by the pediatric surgeons.
  • Most ovarian masses in childhood are benign.
  • Malignant lesions have favorable outcomes with chemotherapy, even with recurrent disease.
  • [MeSH-minor] Adolescent. Child. Child, Preschool. Cohort Studies. Disease-Free Survival. Female. Humans. Incidence. Laparoscopy. Laparotomy. Ovariectomy. Retrospective Studies. Treatment Outcome

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  • (PMID = 19062661.001).
  • [ISSN] 0003-1348
  • [Journal-full-title] The American surgeon
  • [ISO-abbreviation] Am Surg
  • [Language] eng
  • [Publication-type] Journal Article
  • [Publication-country] United States
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28. Mikuz G: [WHO classification of testicular tumors]. Verh Dtsch Ges Pathol; 2002;86:67-75
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  • The most important is obviously the precursor lesion of germ cell tumors, which has been called "intratubular malignant germ cells".
  • Such atypical cells appear in the tubules adjacent to the germ cell tumors, in some few cases (6%) also in the contra lateral healthy gonad and rarely in infertile men (1%).
  • The precursor lesion can progress to franc germ cell tumor starting probably with seminoma, which still maintain the capability of differentiation (pluripotente cells) in all other types of non-seminomatous germ cell tumors.
  • This lesion is missed in germ cell tumors of childhood and in spermatocytic seminomas, both seem to have a histogenetic history rather different from the other germ cell in adults.
  • Unfortunately, however, the old term "teratoma with malignant transformation" was changed to "teratoma with malignant areas" in the 1998 classification.
  • This is a harmless name for an extremely dangerous tumor in which one tissue overgrows the other and gives rise to somatic type sarcomas or carcinomas.
  • Such tumors do not respond like germ cell tumors to the usual chemotherapy.
  • Treatment should be tailored according to that used in standard management of the respective sarcoma or carcinoma.
  • In the comments it is mentioned that the testis carcinoid could be a part of teratoma, but the diagnosis is listed in the group of "miscellaneous" tumors together with tumors of ovarian epithelial type.
  • This is a very questionable decision because the normal testis does not contain neuroendocrine cells from which carcinoids would have to be able to develop.
  • "Large cell calcifying Sertoli cell tumour" has been recently described and can be sporadic or inherited.
  • This morphologically peculiar tumor can be part of the Swiss syndrome also called Carney's complex.
  • The patients have cardiac myxomas, spotty skin pigmentation, hormone active nodular hyperplasia of the adrenals and soft tissue myxomas.
  • The newly appearing "mixed germ cell--sex cord/gonadal stromal tumours, unclassified" has a histology similar to the well known gonadoblastomas.
  • For the therapy of germ cell tumor an assessment of risk factors found by the pathologists is extremely important.
  • The most important independent predictors of relapse are tumor invasion of blood or lymph-vessels, absence of yolk sac elements and the presence of an embryonal carcinoma component.
  • In the absence of such predictors a surveillance policy allows some patients to forgo chemotherapy.

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  • (PMID = 12647353.001).
  • [ISSN] 0070-4113
  • [Journal-full-title] Verhandlungen der Deutschen Gesellschaft für Pathologie
  • [ISO-abbreviation] Verh Dtsch Ges Pathol
  • [Language] ger
  • [Publication-type] English Abstract; Journal Article; Review
  • [Publication-country] Germany
  • [Number-of-references] 48
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