Malignant extracranial germ cell tumours: A first national report by the South African Children's Cancer Study Group

Objective To determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell tumours (MEGCTs) in preparation for the development of a harmonised national treatment protocol. Methods A retrospective folder review was un...

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Veröffentlicht in:	Pediatric blood & cancer Jg. 69; H. 5; S. e29543 - n/a
Hauptverfasser:	Hendricks, Marc, Cois, Annibale, Geel, Jennifer, du Plessis, Johan, Bassingthwaighte, Mairi, Naidu, Gita, Rowe, Biance, Büchner, Ané, Omar, Fareed, Thomas, Karla, Uys, Ronelle, Zyl, Anel, Heerden, Jaques, Mahlachana, Ngoakoana, Vermeulen, Johani, Davidson, Alan, Frazier, A. Lindsay, Donald, Kirsty, Kruger, Mariana
Format:	Journal Article
Sprache:	Englisch
Veröffentlicht:	United States Wiley Subscription Services, Inc 01.05.2022
Schlagworte:	Adolescent alpha-Fetoproteins Antineoplastic Combined Chemotherapy Protocols - therapeutic use Biopsy Chemotherapy Child Children Diagnosis Female Hematology Humans Male malignant extracranial germ cell tumours Medical prognosis Neoplasm Staging Neoplasms, Germ Cell and Embryonal - pathology Oncology paediatric Patients Pediatrics Prognosis Retrospective Studies Sex ratio South Africa - epidemiology Tumors South Africa malignant extracranial germ cell tumours paediatric
ISSN:	1545-5009, 1545-5017, 1545-5017
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Abstract	Objective To determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell tumours (MEGCTs) in preparation for the development of a harmonised national treatment protocol. Methods A retrospective folder review was undertaken at nine South African paediatric oncology units to document patient profiles, tumour and treatment‐related data and outcomes for all children with biopsy‐proven MEGCTs from birth up to and including 16 years of age. Results Between 1 January 2000 and 31 December 2015, 218 patients were diagnosed with MEGCTs. Female sex (hazard ratio [HR] 0.284, p = .037) and higher socio‐economic status (SES) (HR 0.071, p = .039) were associated with a significantly lower risk of death. Advanced clinical stage at diagnosis significantly affected 5‐year OS: stage I: 96%; stage II: 94.3%; stage III: 75.5% (p = .017) and stage IV (60.1%; p < .001). There was a significant association between earlier stage at presentation and higher SES (p = .03). Patients with a serum alpha‐fetoprotein (AFP) level of more than 33,000 ng/ml at diagnosis had significantly poorer outcomes (p = .002). The use of chemotherapy significantly improved survival, irrespective of the regimen used (p < .001). Conclusions The cohort demonstrated a 5‐year OS of 80.3% with an event‐free survival (EFS) of 75.3%. Stage, the use of chemotherapy and an elevated serum AFP level of more than 33,000 ng/ml were independently predictive of outcome. The relationship between SES and outcome is important as the implementation of the new national protocol hopes to standardise care across the socio‐economic divide.
AbstractList	ObjectiveTo determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell tumours (MEGCTs) in preparation for the development of a harmonised national treatment protocol.MethodsA retrospective folder review was undertaken at nine South African paediatric oncology units to document patient profiles, tumour and treatment‐related data and outcomes for all children with biopsy‐proven MEGCTs from birth up to and including 16 years of age.ResultsBetween 1 January 2000 and 31 December 2015, 218 patients were diagnosed with MEGCTs. Female sex (hazard ratio [HR] 0.284, p = .037) and higher socio‐economic status (SES) (HR 0.071, p = .039) were associated with a significantly lower risk of death. Advanced clinical stage at diagnosis significantly affected 5‐year OS: stage I: 96%; stage II: 94.3%; stage III: 75.5% (p = .017) and stage IV (60.1%; p < .001). There was a significant association between earlier stage at presentation and higher SES (p = .03). Patients with a serum alpha‐fetoprotein (AFP) level of more than 33,000 ng/ml at diagnosis had significantly poorer outcomes (p = .002). The use of chemotherapy significantly improved survival, irrespective of the regimen used (p < .001).ConclusionsThe cohort demonstrated a 5‐year OS of 80.3% with an event‐free survival (EFS) of 75.3%. Stage, the use of chemotherapy and an elevated serum AFP level of more than 33,000 ng/ml were independently predictive of outcome. The relationship between SES and outcome is important as the implementation of the new national protocol hopes to standardise care across the socio‐economic divide. To determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell tumours (MEGCTs) in preparation for the development of a harmonised national treatment protocol. A retrospective folder review was undertaken at nine South African paediatric oncology units to document patient profiles, tumour and treatment-related data and outcomes for all children with biopsy-proven MEGCTs from birth up to and including 16 years of age. Between 1 January 2000 and 31 December 2015, 218 patients were diagnosed with MEGCTs. Female sex (hazard ratio [HR] 0.284, p = .037) and higher socio-economic status (SES) (HR 0.071, p = .039) were associated with a significantly lower risk of death. Advanced clinical stage at diagnosis significantly affected 5-year OS: stage I: 96%; stage II: 94.3%; stage III: 75.5% (p = .017) and stage IV (60.1%; p < .001). There was a significant association between earlier stage at presentation and higher SES (p = .03). Patients with a serum alpha-fetoprotein (AFP) level of more than 33,000 ng/ml at diagnosis had significantly poorer outcomes (p = .002). The use of chemotherapy significantly improved survival, irrespective of the regimen used (p < .001). The cohort demonstrated a 5-year OS of 80.3% with an event-free survival (EFS) of 75.3%. Stage, the use of chemotherapy and an elevated serum AFP level of more than 33,000 ng/ml were independently predictive of outcome. The relationship between SES and outcome is important as the implementation of the new national protocol hopes to standardise care across the socio-economic divide. Objective To determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell tumours (MEGCTs) in preparation for the development of a harmonised national treatment protocol. Methods A retrospective folder review was undertaken at nine South African paediatric oncology units to document patient profiles, tumour and treatment‐related data and outcomes for all children with biopsy‐proven MEGCTs from birth up to and including 16 years of age. Results Between 1 January 2000 and 31 December 2015, 218 patients were diagnosed with MEGCTs. Female sex (hazard ratio [HR] 0.284, p = .037) and higher socio‐economic status (SES) (HR 0.071, p = .039) were associated with a significantly lower risk of death. Advanced clinical stage at diagnosis significantly affected 5‐year OS: stage I: 96%; stage II: 94.3%; stage III: 75.5% (p = .017) and stage IV (60.1%; p < .001). There was a significant association between earlier stage at presentation and higher SES (p = .03). Patients with a serum alpha‐fetoprotein (AFP) level of more than 33,000 ng/ml at diagnosis had significantly poorer outcomes (p = .002). The use of chemotherapy significantly improved survival, irrespective of the regimen used (p < .001). Conclusions The cohort demonstrated a 5‐year OS of 80.3% with an event‐free survival (EFS) of 75.3%. Stage, the use of chemotherapy and an elevated serum AFP level of more than 33,000 ng/ml were independently predictive of outcome. The relationship between SES and outcome is important as the implementation of the new national protocol hopes to standardise care across the socio‐economic divide. To determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell tumours (MEGCTs) in preparation for the development of a harmonised national treatment protocol.OBJECTIVETo determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell tumours (MEGCTs) in preparation for the development of a harmonised national treatment protocol.A retrospective folder review was undertaken at nine South African paediatric oncology units to document patient profiles, tumour and treatment-related data and outcomes for all children with biopsy-proven MEGCTs from birth up to and including 16 years of age.METHODSA retrospective folder review was undertaken at nine South African paediatric oncology units to document patient profiles, tumour and treatment-related data and outcomes for all children with biopsy-proven MEGCTs from birth up to and including 16 years of age.Between 1 January 2000 and 31 December 2015, 218 patients were diagnosed with MEGCTs. Female sex (hazard ratio [HR] 0.284, p = .037) and higher socio-economic status (SES) (HR 0.071, p = .039) were associated with a significantly lower risk of death. Advanced clinical stage at diagnosis significantly affected 5-year OS: stage I: 96%; stage II: 94.3%; stage III: 75.5% (p = .017) and stage IV (60.1%; p < .001). There was a significant association between earlier stage at presentation and higher SES (p = .03). Patients with a serum alpha-fetoprotein (AFP) level of more than 33,000 ng/ml at diagnosis had significantly poorer outcomes (p = .002). The use of chemotherapy significantly improved survival, irrespective of the regimen used (p < .001).RESULTSBetween 1 January 2000 and 31 December 2015, 218 patients were diagnosed with MEGCTs. Female sex (hazard ratio [HR] 0.284, p = .037) and higher socio-economic status (SES) (HR 0.071, p = .039) were associated with a significantly lower risk of death. Advanced clinical stage at diagnosis significantly affected 5-year OS: stage I: 96%; stage II: 94.3%; stage III: 75.5% (p = .017) and stage IV (60.1%; p < .001). There was a significant association between earlier stage at presentation and higher SES (p = .03). Patients with a serum alpha-fetoprotein (AFP) level of more than 33,000 ng/ml at diagnosis had significantly poorer outcomes (p = .002). The use of chemotherapy significantly improved survival, irrespective of the regimen used (p < .001).The cohort demonstrated a 5-year OS of 80.3% with an event-free survival (EFS) of 75.3%. Stage, the use of chemotherapy and an elevated serum AFP level of more than 33,000 ng/ml were independently predictive of outcome. The relationship between SES and outcome is important as the implementation of the new national protocol hopes to standardise care across the socio-economic divide.CONCLUSIONSThe cohort demonstrated a 5-year OS of 80.3% with an event-free survival (EFS) of 75.3%. Stage, the use of chemotherapy and an elevated serum AFP level of more than 33,000 ng/ml were independently predictive of outcome. The relationship between SES and outcome is important as the implementation of the new national protocol hopes to standardise care across the socio-economic divide.
Author	Zyl, Anel Hendricks, Marc Vermeulen, Johani Heerden, Jaques Geel, Jennifer Omar, Fareed Frazier, A. Lindsay Cois, Annibale Mahlachana, Ngoakoana Uys, Ronelle Donald, Kirsty Thomas, Karla Rowe, Biance Kruger, Mariana Naidu, Gita Bassingthwaighte, Mairi Büchner, Ané Davidson, Alan du Plessis, Johan
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Notes	SIOP International, Lyon, France (Poster): Hendricks M, Cois A, du Plessis J, Geel J, van Heerden J, Naidu G, Bassingwaighte M, Rowe, B, van Zyl A, Uys R, Büchner A, Omar F, Thomas K, Machlachana N, Vermeulen J, Davidson A, Donald K, Kruger M. Favourable outcomes for children with biopsy‐proven malignant extracranial germ cell tumours 1990–2015: A first national report by the South African Children's Cancer Study Group (SACCSG). Pediatr Blood Cancer. 2019;66(11); SIOP 19‐0822. ObjectType-Article-1 SourceType-Scholarly Journals-1 ObjectType-Feature-2 content type line 14 content type line 23
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Snippet	Objective To determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell... To determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell tumours... ObjectiveTo determine the overall survival (OS) and prognostic factors influencing outcomes in children and adolescents with malignant extracranial germ cell...
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SubjectTerms	Adolescent alpha-Fetoproteins Antineoplastic Combined Chemotherapy Protocols - therapeutic use Biopsy Chemotherapy Child Children Diagnosis Female Hematology Humans Male malignant extracranial germ cell tumours Medical prognosis Neoplasm Staging Neoplasms, Germ Cell and Embryonal - pathology Oncology paediatric Patients Pediatrics Prognosis Retrospective Studies Sex ratio South Africa - epidemiology Tumors
Title	Malignant extracranial germ cell tumours: A first national report by the South African Children's Cancer Study Group
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