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Mortality in Children with Optic Pathway Glioma Treated with Up-Front BB-SFOP Chemotherapy

Rakotonjanahary, Josué ; De Carli, Emilie ; Delion, Matthieu ; Kalifa, Chantal ; Grill, Jacques ; Doz, François ; Leblond, Pierre ; Bertozzi, Anne-Isabelle ; Rialland, Xavier Alonso, Marta M.

PloS one, 2015-06, Vol.10 (6), p.e0127676-e0127676 [Periódico revisado por pares]

United States: Public Library of Science

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Título:
Mortality in Children with Optic Pathway Glioma Treated with Up-Front BB-SFOP Chemotherapy
Autor: Rakotonjanahary, Josué ; De Carli, Emilie ; Delion, Matthieu ; Kalifa, Chantal ; Grill, Jacques ; Doz, François ; Leblond, Pierre ; Bertozzi, Anne-Isabelle ; Rialland, Xavier
Alonso, Marta M.
Assuntos: Age ; Antineoplastic Agents - therapeutic use ; Astrocytoma - drug therapy ; Astrocytoma - mortality ; Brain cancer ; Carboplatin - therapeutic use ; Chemotherapy ; Child ; Child, Preschool ; Children ; Cisplatin - therapeutic use ; Cohort Studies ; Cyclophosphamide - therapeutic use ; Death ; Diagnosis ; Disease-Free Survival ; Drug Therapy, Combination ; Etoposide - therapeutic use ; Evolution ; Female ; France ; Glioma ; Health risk assessment ; Humans ; Hypertension ; Infant ; Male ; Mortality ; Oncology ; Optic Nerve Glioma - drug therapy ; Optic Nerve Glioma - mortality ; Osteoprotegerin ; Patients ; Pediatrics ; Procarbazine - therapeutic use ; Prognosis ; Radiation therapy ; Regression analysis ; Risk analysis ; Risk factors ; Surgery ; Survival ; Survival Rate ; Tumors ; Vincristine - therapeutic use
É parte de: PloS one, 2015-06, Vol.10 (6), p.e0127676-e0127676
Notas: ObjectType-Article-1
SourceType-Scholarly Journals-1
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content type line 23
Membership of the Brain Tumor Committee of SFCE is listed in the Acknowledgments.
Conceived and designed the experiments: JR EDC XR. Performed the experiments: XR EDC MD CK JG FD PL AIB. Analyzed the data: JR EDC XR. Contributed reagents/materials/analysis tools: JR XR. Wrote the paper: JR EDC MD CK JG FD PL AIB XR.
Competing Interests: The authors have declared that no competing interests exist.
Descrição: In terms of overall survival (OS), limited data are available for the very long-term outcomes of children treated for optic pathway glioma (OPG) with up-front chemotherapy. Therefore, we undertook this study with the aim of clarifying long-term OS and causes of death in these patients. We initiated and analyzed a historical cohort study of 180 children with OPG treated in France with BB-SFOP chemotherapy between 1990 and 2004. The survival distributions were estimated using Kaplan-Meier method. The effect of potential risk factors on the risk of death was described using Cox regression analysis. The OS was 95% [95% CI: 90.6-97.3] 5 years after diagnosis and significantly decreased over time without ever stabilizing: 91.6% at 10 years [95% CI: 86.5-94.8], 80.7% at 15 years [95% CI: 72.7-86.8] and 75.5% [95% CI: 65.6-83] at 18 years. Tumor progression was the most common cause of death (65%). Age and intracranial hypertension at diagnosis were significantly associated with a worse prognosis. Risk of death was increased by 3.1[95% CI: 1.5-6.2] (p=0.002) for patients less than 1 year old at diagnosis and by 5.2[95% CI: 1.5-17.6] (p=0.007) for patients with initial intracranial hypertension. Boys without diencephalic syndrome had a better prognosis (HR: 0.3 [95% CI: 0.1-0.8], p=0.007). This study shows that i) in children with OPG, OS is not as favorable as previously described and ii) patients can be classified into 2 groups depending on risk factors (age, intracranial hypertension, sex and diencephalic syndrome) with an OS rate of 50.4% at 18 years [95% CI: 31.4-66.6] in children with the worst prognosis. These findings could justify, depending on the initial risk, a different therapeutic approach to this tumor with more aggressive treatment (especially chemotherapy) in patients with high risk factors.
Editor: United States: Public Library of Science
Idioma: Inglês

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Mortality in Children with Optic Pathway Glioma Treated with Up-Front BB-SFOP Chemotherapy

Rakotonjanahary, Josué ; De Carli, Emilie ; Delion, Matthieu ; Kalifa, Chantal ; Grill, Jacques ; Doz, François ; Leblond, Pierre ; Bertozzi, Anne-Isabelle ; Rialland, Xavier Alonso, Marta M.

United States: Public Library of Science

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