Outcomes After Reirradiation for Recurrent Pediatric Intracranial Ependymoma

Tsang, Derek S.; Burghen, Elizabeth; Klimo, Paul; Boop, Frederick A.; Ellison, David W.; Merchant, Thomas E.

doi:10.1016/j.ijrobp.2017.10.002

Cited by 70 publications

(60 citation statements)

References 21 publications

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“…20 We found both GTR and irradiation improved prognosis, in agreement with previous studies. 2,[21][22][23]36,43 However, only short-term survival benefits were seen, which were limited to combined and PF cohorts, raising questions about the best approach for patients with ST tumors.…”

Section: Discussionmentioning

confidence: 99%

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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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Section: Discussionmentioning

confidence: 99%

“…2,21 Studies suggest that reirradiation of recurrent ependymoma is safe and feasible. [21][22][23] However, there are risks of brainstem radio-necrosis and late effects. Evidence that chemotherapy is beneficial at recurrence is limited to a few small studies.…”

Section: Introductionmentioning

confidence: 99%

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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“…Given that the irradiation dose tolerance of supratentorial brain is higher than that of the brainstem, it is a logical supposition that reirradiation of supratentorial HGG should be similarly safe and effective as reirradiation of brainstem glioma . Reirradiation has also been evaluated in children with intracranial ependymoma and is safe and well‐tolerated, with a low incidence of necrosis …”

Section: Discussionmentioning

confidence: 99%

“…10 Reirradiation has also been evaluated in children with intracranial ependymoma and is safe and well-tolerated, with a low incidence of necrosis. [17][18][19][20] Repeat irradiation also has an emerging role in adults with recurrent glioblastoma. 9 Strengths of this study include the assembly of the largest known cohort of children with recurrent HGG treated with a repeat course of RT, with a comparison to a group of children who were not reirradiated.…”

Section: Prior Studiesmentioning

confidence: 99%

Repeat irradiation for children with supratentorial high‐grade glioma

Tsang

Oliveira

Bouffet

et al. 2019

Pediatric Blood & Cancer

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Background There are very few studies about the role of repeat irradiation (RT2) for children with recurrent supratentorial high‐grade glioma (HGG). It was the aim of this study to assess the effectiveness and safety of RT2 in this population. Procedure This was a retrospective cohort study of 40 children age 18 years and under with recurrent supratentorial HGG who had received at least one course of RT. In‐field reirradiation volumes included focal or whole brain RT, with doses ranging from 30 to 54 Gy. The primary endpoint was overall survival (OS) from the first day of RT2. Results Fourteen patients underwent RT2. The median survival of these patients was 6.5 months. Patients with ≥12 months elapsed time between RT1 and RT2 experienced longer OS than patients who had < 12 months (P = 0.009). There was no difference in OS between patients with or without germline mutations (e.g., Lynch, Li–Fraumeni, or constitutional mismatch‐repair deficiency, P = 0.20). Ten patients received RT2 that overlapped with RT1 volumes for locally recurrent disease. Of this group, 80% experienced clinical benefit from in‐field RT2, defined as clinical/radiologic response or stable disease. Ninety‐three percent completed the prescribed course of RT2, with one patient developing grade 3 radiation necrosis four months after RT2. When compared with 26 patients who were not offered reirradiation, those selected for RT2 had improved median survival from the time of first disease progression (9.4 vs 3.8 months, P = 0.005). Conclusions Reirradiation for children with recurrent supratentorial HGG is a safe, effective treatment that provides short‐term disease control.

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“…Reirradiation could increase the time of survival, and amelioration of quality of life, which are very important factors for patients and parents. Even a third course of radiation therapy had been reported Tsang D et al 25 and may be helpful in selected cases of ependymomas, for local control and palliation.…”

Section: Discussionmentioning

confidence: 99%

Reirradiation of Recurrent Tumors in Central Nervous System in Children and Adolescents

Alert¹,

Chon²,

Ropero³

2019

Neuro Open J

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I n Cuba, tumors of the Central Nervous System (CNS) for children and adolescents account between 18 and 20% of all tumors in this group of age. 1-3 The main methods of treatment are surgery, radiotherapy, and chemotherapy. 4 Radiation therapy is a major treatment avenue in medulloblastomas, primitive neuroectodermal tumor (PNET) in some cases of germinomas consist of craneospinal irradiation (CS), and a supplementary boost to the post-operative tumor bed, followed by chemotherapy. In

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Outcomes After Reirradiation for Recurrent Pediatric Intracranial Ependymoma

Cited by 70 publications

References 21 publications

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

Repeat irradiation for children with supratentorial high‐grade glioma

Reirradiation of Recurrent Tumors in Central Nervous System in Children and Adolescents

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