Pediatric Localized Intracranial Ependymomas: A Multicenter Analysis of the Société Française de lutte contre les Cancers de l'Enfant (SFCE) from 2000 to 2013

Ducassou, A.; Padovani, Laëtitia; Chaltiel, Léonor; Bolle, Stéphanie; Habrand, Jean‐Louis; Claude, L.; Carrié, C.; Muracciole, Xavier; Coche‐Dequeant, B.; Alapetite, Claire; Supiot, S.; Demoor-Goldschmidt, Charlotte; Bernier-Chastagner, Valérie; Huchet, A.; Leseur, J.; Prisé, É. Le; Kerr, Christine; Truc, G.; Nguyen, Tan Dat; Bertozzi, Anne‐Isabelle; Frappaz, Didier; Boetto, S.; Sévely, A; Tensaouti, F.; Laprie, Anne

doi:10.1016/j.ijrobp.2018.05.036

Cited by 22 publications

(12 citation statements)

References 25 publications

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“…Furthermore, in spite of extensive investigation and research, the role of chemotherapy in the management of EPN remains unclear. Consistent with our results, several study cohorts of pediatric or adult patients in which the effect of chemotherapy was retrospectively analyzed also failed to demonstrate a survival advantage . Nevertheless, this modality of treatment might be beneficial to some subsets of EPN patients with the ongoing therapeutic challenges.…”

Section: Discussionsupporting

confidence: 87%

Personalizing age‐specific survival prediction and risk stratification in intracranial grade II/III ependymoma

et al. 2019

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Background: Models for estimation of survival rates of patients with intracranial grade II/III ependymoma (EPN) are scarce. Considering the heterogeneity in prognostic factors between pediatric and adult patients, we aimed to develop age-specific nomograms for predicting 3-, 5-, and 8-year survival for these patients. Methods: A total of 1390 cases (667 children; 723 adults) of intracranial grade II/ III EPNs diagnosed between 1988 and 2015 were extracted from the Surveillance, Epidemiology, and End Results (SEER) database for our study. Univariable and multivariable Cox analyses were employed to identify independent prognostic predictors. Age-specific nomograms were developed based on the results of multivariate Cox analyses. We also evaluated the performance of these predictive models by concordance index, calibration curves, time-dependent receiver operating characteristic curves, and decision curve analyses. Results: Considerable heterogeneity in prognostic factors was highlighted between pediatric and adult patients. Age, sex, tumor grade, surgery treatment and radiotherapy were identified as significant predictors of overall survival for children, and age, tumor grade, tumor size, surgery treatment, and marital status for adult. Based on these factors, age-specific nomogram models were established and internally validated. These models exhibited favorable discrimination and calibration characteristics. Nomogram-based risk classification systems were also constructed to facilitate risk stratification in EPNs for optimization of clinical management. Conclusions:We developed the first nomograms and corresponding risk classification systems for predicting survival in patients with intracranial grade II/III EPN.These easily used tools can assist oncologists in making accurate survival evaluation. K E Y W O R D Sgrade II/III ependymoma, intracranial, nomogram, overall survival, SEER 616 | DENG Et al.

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Section: Discussionsupporting

confidence: 87%

Personalizing age‐specific survival prediction and risk stratification in intracranial grade II/III ependymoma

et al. 2019

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“…Sixty-two percent of cases relapsed, with 57% five-year cumulative incidence and no difference between intracranial locations. This was higher than two recent studies, 42,43 although all patients were irradiated at primary diagnosis compared with 45% in our cohort. For irra-diated patients, we found a similar relapse incidence to Ducassou 42 (38% vs 31.3% for local relapses) but the incidence was still higher than Merchant 43 (16.2% for local relapses).…”

Section: Discussioncontrasting

confidence: 73%

A retrospective analysis of recurrent pediatric ependymoma reveals extremely poor survival and ineffectiveness of current treatments across central nervous system locations and molecular subgroups

Ritzmann

Rogers

Paine

et al. 2020

Pediatric Blood & Cancer

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Background Relapse occurs in 50% of pediatric ependymoma cases and has poor prognosis. Few studies have investigated the clinical progress of relapsed disease, and treatment lacks a standardized approach. Methods and materials We analyzed 302 pediatric ependymoma cases. Tumor, demographic, and treatment variables were investigated for association with relapse risk, time to recurrence, and survival after relapse. DNA methylation profiling was performed for 135/302 cases, and predominant subgroups were EPN_PFA (n = 95) and EPN_RELA (n = 24). Chromosome 1q status was ascertained for 185/302 cases by fluorescent in‐situ hybridization (FISH), multiplex ligation‐dependent probe amplification (MLPA), and DNA methylation profiles. Results Sixty‐two percent of cases relapsed, with a median of two recurrences with no difference between posterior fossa and supratentorial locations (66% vs 55% relapse rate). One hundred seventeen (38%) cases relapsed within two years and five (2%) beyond 10 years. The late relapses were clinically heterogeneous. Tumor grade and treatment affected risk and time to relapse variably across subgroups. After relapse, surgery and irradiation delayed disease progression with a minimal impact on survival across the entire cohort. In the EPN_PFA and EPN_RELA groups, 1q gain was independently associated with relapse risk (subhazard ratio [SHR] 4.307, P = 0.027 and SHR 1.982, P = 0.010, respectively) while EPN_PFA had increased relapse risk compared with EPN_RELA (SHR = 0.394, P = 0.018). Conclusions Recurrent pediatric ependymoma is an aggressive disease with poor outcomes, for which current treatments are inadequate. We report that chromosome 1q gain increases relapse risk in common molecular subgroups in children but a deeper understanding of the underlying biology at relapse and novel therapeutic approaches are urgently needed.

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“…Gy in 33 fractions) improves clinical outcome with acceptable toxicity even in children younger than 3 years(17)(18)(19)(20)(21). A dose modification to 54 Gy in 30 fractions is recommended in very young children (<12 months) or those undergoing multiple surgeries for tumours near the brainstem because of possible increased risk of brainstem toxicity in these patient groups[45][46][47][48] (Level I). o Radiotherapy should ideally start within 6 weeks of surgery (Level III) o There is no proven role for the use of chemotherapy to delay radiotherapy.…”

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confidence: 99%

A rapid review of evidence and recommendations from the SIOPE radiation oncology working group to help mitigate for reduced paediatric radiotherapy capacity during the COVID-19 pandemic or other crises

Janssens

Mandeville

Timmermann

et al. 2020

Radiotherapy and Oncology

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Objective: To derive evidence-based recommendations for the optimal utilisation of resources during unexpected shortage of radiotherapy capacity. Methods and materials: We have undertaken a rapid review of published literature on the role of radiotherapy in the multimodality treatment of paediatric cancers governing the European practise of paediatric radiotherapy. The derived data has been discussed with expert paediatric radiation oncologists to derive a hierarchy of recommendations. Results: The general recommendations to mitigate the potential detriment of an unexpected shortage of radiotherapy facilities include: (1) maintain current standards of care as long as possible (2) refer to another specialist paediatric radiotherapy department with similar level of expertise (3) prioritise use of existing radiotherapy resources to treat patients with tumours where radiotherapy has the most effect on clinical outcome (4) use chemotherapy to defer the start of radiotherapy where timing of radiotherapy is not expected to be detrimental (5) active surveillance for low-grade tumours if appropriate and (6) consider iso-effective hypofractionated radiotherapy regimens only for selected patients with predicted poor prognosis. The effectiveness of radiotherapy and recommendations for prioritisation of its use for common and challenging paediatric tumours are discussed. Conclusion:This review provides evidence-based treatment recommendations during unexpected shortage of paediatric radiotherapy facilities. It has wider applications for the optimal utilisation of facilities, to improve clinical outcome in low-and middle-income countries, where limited resources continue to be a challenge.

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Pediatric Localized Intracranial Ependymomas: A Multicenter Analysis of the Société Française de lutte contre les Cancers de l'Enfant (SFCE) from 2000 to 2013

Cited by 22 publications

References 25 publications

Personalizing age‐specific survival prediction and risk stratification in intracranial grade II/III ependymoma

Personalizing age‐specific survival prediction and risk stratification in intracranial grade II/III ependymoma

A retrospective analysis of recurrent pediatric ependymoma reveals extremely poor survival and ineffectiveness of current treatments across central nervous system locations and molecular subgroups

A rapid review of evidence and recommendations from the SIOPE radiation oncology working group to help mitigate for reduced paediatric radiotherapy capacity during the COVID-19 pandemic or other crises

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