Mediastinal Germ Cell Tumors in Pediatric Patients: A Report From the Italian Association of Pediatric Hematology and Oncology

Pasquale, Maria Debora De; Crocoli, Alessandro; Conte, Massimo; Indolfi, Paolo; D’Angelo, Paolo; Boldrini, Renata; Terenziani, Monica; Inserra, Alessandro

doi:10.1002/pbc.25895

Cited by 15 publications

(22 citation statements)

References 20 publications

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“…Diagnosis and histology were performed according to the World Health Organization (WHO) classification. 31 Immunohistochemistry and manual acquisition IHC was conducted with 4 μm of formaldehyde-fixed paraffinembedded tissues sections. 24 Following deparaffinization and antigen retrieval with EnVision FLEX Target retrieval solution at low or high pH (citrate buffer pH 6.1 and Tris/EDTA pH 9.0, respectively) with PT-link (Dako), slides were subjected to the avidin/ biotin blocking system (Thermo Fisher Scientific, Fremont, CA, USA) according to manufacturer's instructions.…”

Section: Patientsmentioning

confidence: 99%

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Tumor-infiltrating T cells and PD-L1 expression in childhood malignant extracranial germ-cell tumors

et al. 2018

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Although pediatric malignant extracranial germ-cell tumors (meGCTs) are among the most chemosensitive solid tumors, a group of patients relapse and die of disease. To identify new markers predicting clinical outcome, we examined the prognostic relevance of tumor-infiltrating T lymphocytes (TILs) and the expression of PD-1 and PD-L1 in a cohort of pediatric meGCTs by in situ immunohistochemistry. MeGCTs were variously infiltrated by T cell-subtypes according to the tumor subtype, tumor location and age at diagnosis. We distinguished three different phenotypes: i) tumors not infiltrated by T cells (immature teratomas and half of the yolk sac tumors), ii) tumors highly infiltrated by CD8 + T cells expressing PD-1, which identifies activated tumor-reactive T cells (seminomas and dysgerminomas), iii) tumors highly infiltrated by CD8 + T cells within an immunosuppressive tumor microenvironment characterized by CD4 + FOXP3 + Treg cells and PD-L1-expressing tumor cells (embryonal carcinomas, choriocarcinomas and the remaining yolk sac tumors). Tumor subtypes belonging mixed meGCTs were variously infiltrated, suggesting the coexistence of multiple immune microenvironments either facilitating or precluding the entry of T cells.These findings support the hypothesis that TILs influence the development of meGCTs and might be of clinical relevance to improve risk stratification and the treatment of pediatric patients. ARTICLE HISTORY

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Section: Patientsmentioning

confidence: 99%

“…28 Although the majority of patients are cured by surgery and chemotherapy, a subgroup of them relapse. [29][30][31][32][33] Current therapeutic options are effective in less than 50% of patients with high-risk disease, indicating that more effective and less toxic therapies are urgently needed.…”

Section: Introductionmentioning

confidence: 99%

Tumor-infiltrating T cells and PD-L1 expression in childhood malignant extracranial germ-cell tumors

et al. 2018

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“…In addition, most patients with high-risk malignant tumors chose to go to well-known hospital for better treatment, especially in our city. In De Pasquale et al [19] study, 70% patients with malignant mediastinal GCTs had stage III, and the remaining patients had stage IV with distant metastases.…”

Section: Discussionmentioning

confidence: 99%

Gonadal germ cell tumors in children

Lin

Zheng

et al. 2017

Medicine

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Background:The true incidence of gonadal germ cell tumors (GCTs) in children is unknown. Few studies have been published concerning about pediatric gonadal GCTs. The aim of this study is to review and analyze clinical data on the diagnosis and management of gonadal GCTs in children.Methods:Between 2005 and 2015, 127 pediatric patients (<14 years old) with gonadal GCTs admitted to our institute were reviewed. Clinical features, imaging and laboratory studies, surgical approaches, as well as pathological diagnoses were recorded.Results:The series comprised 53 males with testicular GCTs and 74 females with ovarian GCTs. Their median age was 5.8 years old. Palpable mass was the main clinical manifestation of testicular GCTs, while abdominal pain and abdominal distention were the most frequent presenting symptoms of ovarian GCTs. Both computed tomography and magnetic resonance imaging showed a high diagnostic yield. AFP levels were elevated in most malignant GCTs, markedly elevated in yolk sac tumors. All patients were treated surgically. Mature teratoma was the most common type of benign GCTs, while yolk sac tumor was the most common type of malignant GCTs.Conclusion:Gonadal GCTs in children have various of pathological types, as well as clinical manifestations. Imaging and laboratory data could be useful for differentiation of malignant from benign tumors. Final diagnosis depends on pathology. Surgical excision of the gonadal GCTs is the prior option.

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“…The mediastinum is the most common extragonadal primary tumor site[ 4 ]. Mediastinal germ cell tumors (MGCTs) account for 19%–25% of all mediastinal tumors and 3%–6% of all GCTs in pediatric age[ 1 , 2 , 3 , 19 , 20 ]. In our experience, the mediastinal site represents 7.26% of all GCTs registered from 2005 to 2014 at Sun Yat-Sen University Cancer Center.…”

Section: Discussionmentioning

confidence: 99%

“…The mediastinal germ cell tumors (GCTs) are rare in children and adolescents, comprising 3–6% of all GCTs diagnosed among children and adolescents younger than 18 years of age[ 1 , 2 , 3 ]. As the most common extragonadal GCTs (EGCTs), these tumors are considered to arise from the abnormal migration of germ cells during embryogenesis[ 4 ].…”

Section: Introductionmentioning

confidence: 99%

Role of post-chemotherapy radiation in the management of children and adolescents with primary advanced malignant mediastinal germ cell tumors

et al. 2017

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ObjectivePrimary malignant mediastinal germ cell tumors (MMGCTs) are rare in children and adolescents and have a poorer prognosis than their gonadal counterparts. We report a single institutional experience of a 10-year period of primary advanced MMGCTs treated with chemotherapy, followed by radiotherapy in those who had residual mass.MethodsChildren and adolescents with primary advanced MMGCTs between 2005 and 2014 were identified from the Cancer Center, Sun Yat-Sen University. Medical records were reviewed for clinicopathological characteristics, treatments, and outcomes.ResultsTwenty-four children and adolescents with either stage III or IV primary advanced MMGCTs met the inclusion criteria. There were 23 males and one female with a median age of 16 (range 10–18). Seven cases were seminomas (29.2%); four (16.7%) yolk sac tumors (YST); three (12.5%) choriocarcinomas; and ten (41.6%) nonteratomatous combined germ cell tumors (CGCTs). All patients were treated with first-line cisplatin-based chemotherapy regimens (PEB: 19, VIP: 5). Thirteen (54.2%) and Twelve (50%) patients received surgery and radiotherapy, respectively. With a median follow-up of 46.2 months (range 9.6–124.8 months), a total of five (20%) patients died of disease progression; the five-year overall survival (OS) and disease-free survival (DFS) rates were 82.3% and 64.9%, respectively.—Seven patients with seminoma GCTs received post-chemotherapy irradiation were alive with sustained CR (5-year OS and DFS, 100%, respectively). Five patients with NSGCTs were administered irradiation and one relapsed 35 months later and died of metastasis (5-year OS, 100%; 5-year DFS 66.7%). Univariate analysis identified histology and stage were prognostic factors.ConclusionMultimodality treatment approach of chemotherapy followed by radiation consolidation ensured long-term survival in primary advanced MMGCTs. Further research is warranted to improve the prognosis of children with primary advanced MMGCTs.

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Mediastinal Germ Cell Tumors in Pediatric Patients: A Report From the Italian Association of Pediatric Hematology and Oncology

Cited by 15 publications

References 20 publications

Tumor-infiltrating T cells and PD-L1 expression in childhood malignant extracranial germ-cell tumors

Tumor-infiltrating T cells and PD-L1 expression in childhood malignant extracranial germ-cell tumors

Gonadal germ cell tumors in children

Role of post-chemotherapy radiation in the management of children and adolescents with primary advanced malignant mediastinal germ cell tumors

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