Laetitia Callec1, Audrey Lardy-Cleaud2, Lea Guerrini-Rousseau3, Claire Alapetite4, Laure Vignon5, Pascal Chastagner6, Didier Frappaz7, Cecile Faure-Conter8. 1. Laetitia Callec, CHU- Hôpitaux de Brabois, Department of Pediatrics, Vandoeuvre les Nancy, France. 2. Audrey Lardy-Cleaud, Department of Biostatistic, Centre Léon Bérard, Lyon, France. 3. Lea Guerrini-Rousseau, Department of Pediatric and Adolescents Oncology, Institut Gustave Roussy, Paris-Saclay University, Villejuif, France. 4. Claire Alapetite, Institut Curie, Department of Radiation Oncology, Paris, France. 5. Lucie Vignon, INSERM, Paris-Descartes University, Department of Epidemiology, French National Registry of Childhood Solid Tumour Registry (RNTSE), Villejuif, France. 6. Pascal Chastagner, CHU- Hôpitaux de Brabois, Department of Pediatrics, Vandoeuvre les Nancy, France. 7. Didier Frappaz, Institut d'Hemato-oncologie Pediatrique, Lyon, France. 8. Cecile Faure-Conter, Institut d'Hemato-oncologie Pediatrique, Lyon, France. Electronic address: cecile.conter@ihope.fr.
Abstract
BACKGROUND: The optimal therapeutic strategy for relapsing intracranial germ cell tumours (IGCTs) has not been clearly established. METHODS: Relapses of IGCTs, occurring from 01/01/1990 to 31/12/2014, were retrieved from the Societe Française d'Oncologie Pediatrique-TGM 90, 92 and GCT 96 protocols, and from the National Childhood Solid Tumour Registry. Refractory IGCTs were excluded. RESULTS: Forty-four relapsing IGCTs were identified: 14 were initially treated for histologically proven germinomas (germinoma group), 5 for non-histologically proven germinomas (putative germinoma group) and 25 for non-germinomatous germ cell tumours (NGGCTs) (NGGCT group). In the germinoma group, the 5-year event-free survival (EFS) and overall survival (OS) were 79% (95% confidence interval [CI]: 47-93) and 86% (95% CI: 54-96), respectively. Only one of the 11 patients treated with reirradiation experienced a further relapse. A trend of better EFS was observed for relapses at sites that were not initially involved: 5-year EFS of 100% versus 67% (95% CI: 28-88), p = 0.09. In the putative germinoma group, 4 of 5 patients experienced a further event, leading to 2 deaths. In the NGGCT group, the 5-year EFS and OS were 56% (95% CI: 35-73) and 60% (95% CI: 38-76), respectively. A significant improvement in outcomes after high-dose chemotherapy (HDC) was observed: 5-year OS of 72% (95% CI: 46-87) versus 29% (95% CI: 4-61), p = 0.006. CONCLUSION: Relapsing germinomas are highly curable; reirradiation appears to play a key role. Histological proof at initial diagnosis if markers are negative is crucial. Despite inferior outcomes relapsing, NGGCTs can be cured in a significant proportion of cases provided intensive treatment including HDC is applied.
BACKGROUND: The optimal therapeutic strategy for relapsing intracranial germ cell tumours (IGCTs) has not been clearly established. METHODS: Relapses of IGCTs, occurring from 01/01/1990 to 31/12/2014, were retrieved from the Societe Française d'Oncologie Pediatrique-TGM 90, 92 and GCT 96 protocols, and from the National Childhood Solid Tumour Registry. Refractory IGCTs were excluded. RESULTS: Forty-four relapsing IGCTs were identified: 14 were initially treated for histologically proven germinomas (germinoma group), 5 for non-histologically proven germinomas (putative germinoma group) and 25 for non-germinomatous germ cell tumours (NGGCTs) (NGGCT group). In the germinoma group, the 5-year event-free survival (EFS) and overall survival (OS) were 79% (95% confidence interval [CI]: 47-93) and 86% (95% CI: 54-96), respectively. Only one of the 11 patients treated with reirradiation experienced a further relapse. A trend of better EFS was observed for relapses at sites that were not initially involved: 5-year EFS of 100% versus 67% (95% CI: 28-88), p = 0.09. In the putative germinoma group, 4 of 5 patients experienced a further event, leading to 2 deaths. In the NGGCT group, the 5-year EFS and OS were 56% (95% CI: 35-73) and 60% (95% CI: 38-76), respectively. A significant improvement in outcomes after high-dose chemotherapy (HDC) was observed: 5-year OS of 72% (95% CI: 46-87) versus 29% (95% CI: 4-61), p = 0.006. CONCLUSION: Relapsing germinomas are highly curable; reirradiation appears to play a key role. Histological proof at initial diagnosis if markers are negative is crucial. Despite inferior outcomes relapsing, NGGCTs can be cured in a significant proportion of cases provided intensive treatment including HDC is applied.
Authors: Matthew J Murray; Rafael Moleron; Jennifer Adamski; Martin English; G A Amos Burke; Justin Cross; Thankamma Ajithkumar; Sara Stoneham; James C Nicholson Journal: Pediatr Blood Cancer Date: 2021-09-14 Impact factor: 3.167
Authors: Didier Frappaz; Girish Dhall; Matthew J Murray; Stuart Goldman; Cecile Faure Conter; Jeffrey Allen; Rolf Dieter Kortmann; Daphne Haas-Kogen; Giovanni Morana; Jonathan Finlay; James C Nicholson; Ute Bartels; Mark Souweidane; Stefan Schönberger; Alexandre Vasiljevic; Patricia Robertson; Assunta Albanese; Claire Alapetite; Thomas Czech; Chin C Lau; Patrick Wen; David Schiff; Dennis Shaw; Gabriele Calaminus; Eric Bouffet Journal: Neuro Oncol Date: 2022-04-01 Impact factor: 13.029