Outcomes of salvage re-irradiation in recurrent medulloblastoma correlate with age at initial diagnosis, primary risk-stratification, and molecular subgrouping

Gupta, Tejpal; Maitre, Priyamvada; Sastri, Goda Jayant; Krishnatry, Rahul; Shirsat, Neelam; Epari, Sridhar; Sahay, Ayushi; Chinnaswamy, Girish; Patil, Vijay; Shetty, Prakash; Moiyadi, Aliasgar

doi:10.1007/s11060-019-03225-9

Cited by 30 publications

(36 citation statements)

References 28 publications

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“…Furthermore, Sick Children Hospital experience showed that re-irradiation can offer some patients disease control, particularly those with focally recurrent disease in the brain but poor outcome in symptomatic recurrence or disseminated disease (13). Recently, Gupta et al provided encouraging survival outcomes with acceptable toxicity in selected recurrent medulloblastoma treated by reirradiation as part of multimodality salvage therapy (14).…”

Section: Introductionmentioning

confidence: 99%

“…Craniospinal irradiation (CSI) as a component of re-irradiation was previously reported in small series (12,13,14). Recently, the Sick Children group suggested that CSI could be considered one of the treatment tools to help with distant tumor control, considering the very high incidence of distant failures after focal reirradiation (13).…”

Section: Introductionmentioning

confidence: 99%

“…Recently, the Sick Children group suggested that CSI could be considered one of the treatment tools to help with distant tumor control, considering the very high incidence of distant failures after focal reirradiation (13). Moreover, Gupta et al suggested that recurrence medulloblastoma with leptomeningeal dissemination could be bene ted by re-CSI (14).…”

Section: Introductionmentioning

confidence: 99%

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Craniospinal irradiation as part of re-irradiation for children with recurrent medulloblastoma

et al. 2021

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BackgroundMany studies have demonstrated in the last years that once medulloblastoma has recurred, the probability of regaining tumor control is poor despite salvage therapy. Although re-irradiation has an emerging role in other relapsed brain tumors, there is a lack of strong data on re-irradiation for medulloblastoma. MethodsThis is a retrospective cohort study of patients aged 18 years or under, treated at least by a second course of external beam for recurrence medulloblastoma at Garrahan Hospital between 2009 and 2020. Twenty-four patients met eligibility criteria for inclusion. All patients received upfront radiotherapy as part of the curative-intent rst radiotherapy, either craniospinal irradiation (CSI) followed by posterior fossa boost in 20 patients or focal posterior fossa radiation in 4 infants. The second course of radiation consisted of CSI in 15 and focal in 9. The 3-year post rst failure OS (50% vs. 0%; p = 0.0010) was signi cantly better for children who received re-CSI compared to children who received focal re-irradiation. Similarly, the 3-year post-re-RT PFS (31% vs. 0%; p = 0.0005) and OS (25% vs. 0%; p = 0.0003) was signi cantly improved for patients who received re-CSI compared to patients who received focal re-irradiation. No symptomatic intratumoral haemorrhagic events or symptomatic radionecrosis were observed. Survivors fell within mild to moderate intellectual disability range, with a median IQ at last assessment of 58 (range 43-69). ConclusionRe-irradiation with CSI is a safe and effective treatment for children with relapsed medulloblastoma; improves disease control and survival compared with focal re-irradiation. However this approach carries a high neurocognitive cost.All radiation treatments were given at Garrahan Hospital, Buenos Aires, Argentina. Photon external beam therapy was used for all patients. All but four patients received CSI as part of the rst radiation course (RT1), followed by a boost to the entire posterior fossa. Standard risk (SR) patients received CSI 23.4Gy followed by posterior fossa boost 30.6Gy and high-risk (HR) patients received CSI 36Gy followed by posterior fossa boost 19.8Gy. Except for one, all of them received maintenance platinum based chemotherapy; SR as per ACNS0331 (N=2) and COG 9961 (regimen A=3, regimen B= 6); HR as per ACNS0332 (Regimen A=4, Regimen B=4). Four patients received upfront posterior fossa radiotherapy (54 Gy) due to the young age at diagnosis as per standard of care treatment administered between 2002 and 2020 at Hospital Garrahan, based on a modi ed POG-9934 strategy (15). Upon recurrence, most patients with brain solitary lesions were offered surgery followed by metronomic chemotherapy and a second course of irradiation (RT2), while those with multifocal disease received chemotherapy followed by radiotherapy. CSI was administered using standard beam's eye-view treatment planning techniques. Boost treatment and focal radiotherapy was administered using 3D-conformal radiation therapy methods. Hypofractionated stereotactic radiother...

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Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Craniospinal irradiation as part of re-irradiation for children with recurrent medulloblastoma

et al. 2021

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“…The evaluation of all these factors focus the need for a careful patient selection process; this is also confirmed by a recent series published by Gupta et al concerning 28 patients with relapsed medulloblastoma who underwent salvage reirradiation, performed either with re-craniospinal RT or with focal treatments both in conventional or hyperfractionated regimens (32). The authors reported an acceptable profile of toxicity, describing only one case of symptomatic late radionecrosis after re-craniospinal irradiation.…”

Section: Discussionmentioning

confidence: 81%

“…External-beam radiotherapy can be offered either in a definitive setting or after maximal safe resection, despite most of the literature reports data concerning exclusive intent. To date, almost 300 cases of re-irradiation after craniospinal radiotherapy are reported in the literature (Table I), but it is hard to compare these data due to the heterogeneity of the in vivo 34: 1283-1288 (2020) cohorts; furthermore, different RT schedules are used with or without chemotherapy, and only a few reports are available on the use of stereotactic radiotherapy (17)(18)(19)(20)(21)(22)(23)(24)(25)(26)(27)(28)(29)(30)(31)(32).…”

Section: Discussionmentioning

confidence: 99%

Salvage Re-irradiation Options in Adult Medulloblastoma: A Case Report and Review of the Literature

Cuccia

Mortellaro²,

Ognibene³

et al. 2020

In Vivo

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Background/Aim: Medulloblastoma is a rare tumor of adult age, while it occurs more frequently in children. Given the rarity, there is a lack of evidence for the treatment of recurrent disease. Few data are available about salvage reirradiation collecting very heterogeneous series. Case Report: A 51-year-old male presented with headache, nausea, double vision, and gait disorders. A contrast-enhanced brain-MRI showed the presence of multifocal medulloblastoma. After surgery, adjuvant craniospinal radiotherapy was performed, chemotherapy was stopped due to toxicity. After 27 months, a new MRI and a Methionine-PET revealed a late pontocerebellar relapse; multidisciplinary board decided for a SBRT treatment. The second course of RT was well tolerated and 14 months later, the patient is alive in good general conditions, with no evidence of disease. Conclusion: Our experience supports the use of salvage stereotactic radiotherapy as a safe and effective treatment option. Medulloblastoma is one of the most common central nervous system tumors of childhood (1), while it has an extremely lower incidence in the adult age, particularly in >40 years patients (2). Furthermore, in older patients it may occur with unusual presentations, like multifocal disease, of which only six cases are described in the literature (3-8). Due to its rarity in the older 1283 This article is freely accessible online.

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Early outcome after craniospinal irradiation with pencil beam scanning proton therapy for children, adolescents and young adults with brain tumors

Vazquez

Bachmann

Pica

et al. 2022

Pediatric Blood & Cancer

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Central nervous system (CNS) tumors are the most common solid malignancies in children and adolescents and young adults (C-AYAs). Craniospinal irradiation (CSI) is an essential treatment component for some malignancies, but it can also lead to important toxicity. Pencil beam scanning proton therapy (PBSPT) allows for a minimization of dose delivered to organs at risk and, thus, potentially reduced acute and late toxicity. This study aims to report the clinical outcomes and toxicity rates after CSI for C-AYAs treated with PBSPT. Seventy-one C-AYAs (median age: 7.4 years) with CNS tumors were treated with CSI between 2004 and 2021. Medulloblastoma (n = 42: 59%) and ependymoma (n = 8; 11%) were the most common histologies. Median prescribed total PBSPT dose was 54 Gy RBE (range: 18-60.4), and median prescribed craniospinal dose was 24 Gy RBE (range: 18-36.8). Acute and late toxicities were coded according to Common Terminology Criteria for Adverse Events. After a median follow-up of 24.5 months, the estimated 2-year local control, distant control, and overall survival were 86.3%, 80.5%, and 84.7%, respectively. Late grade ≥3 toxicity-free rate was 92.6% at 2 years. Recurrent and metastatic tumors were associated with worse outcome. In conclusion, excellent tumor control with low toxicity rates was observed in C-AYAs with brain tumors treated with CSI using PBSPT. K E Y W O R D Sadolescents and young adults, brain tumors, children, patient-related outcomes, pencil beam scanning, proton therapy, toxicity INTRODUCTIONCraniospinal irradiation (CSI) is an essential treatment component along with surgery and chemotherapy for many children, adolescents

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Outcomes of salvage re-irradiation in recurrent medulloblastoma correlate with age at initial diagnosis, primary risk-stratification, and molecular subgrouping

Cited by 30 publications

References 28 publications

Craniospinal irradiation as part of re-irradiation for children with recurrent medulloblastoma

Craniospinal irradiation as part of re-irradiation for children with recurrent medulloblastoma

Salvage Re-irradiation Options in Adult Medulloblastoma: A Case Report and Review of the Literature

Early outcome after craniospinal irradiation with pencil beam scanning proton therapy for children, adolescents and young adults with brain tumors

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