Suggestions for Escaping the Dark Ages for Pediatric Diffuse Intrinsic Pontine Glioma Treated with Radiotherapy: Analysis of Prognostic Factors from the National Multicenter Study
2
조회
0
다운로드
1
인용
0
참고문헌
키워드
초록
PurposeThis multicenter retrospective study aimed to investigate clinical, radiologic, and treatment-related factors affecting survival in patients with newly diagnosed diffuse intrinsic pontine glioma (DIPG) treated with radiotherapy.Materials and MethodsPatients aged <30 years who underwent radiotherapy as an initial treatment for DIPG between 2000 and 2018 were included; patients who did not undergo magnetic resonance imaging at diagnosis and those with pathologically diagnosed grade I glioma were excluded. We examined medical records of 162 patients collected from 10 participating centers in Korea. The patients’ clinical, radiological, molecular, and histopathologic characteristics, and treatment responses were evaluated to identify the prognosticators for DIPG and estimate survival outcomes.ResultsThe median follow-up period was 10.8 months (interquartile range, 7.5 to 18.1). The 1- and 2-year overall survival (OS) rates were 53.5% and 19.0%, respectively, with a median OS of 13.1 months. Long-term survival rate (≥ 2 years) was 16.7%, and median OS was 43.6 months. Age (< 10 years), poor performance status, treatment before 2010, and post-radiotherapy necrosis were independently associated with poor OS in multivariate analysis. In patients with increased post-radiotherapy necrosis, the median OS estimates were 13.3 months and 11.4 months with and without bevacizumab, respectively (p=0.138).ConclusionTherapeutic strategy for DIPG has remained unchanged over time, and the associated prognosis remains poor. Our findings suggest that appropriate efforts are needed to reduce the occurrence of post-radiotherapy necrosis. Further well-designed clinical trials are recommended to improve the poor prognosis observed in DIPG patients.
Abstract
PurposeThis multicenter retrospective study aimed to investigate clinical, radiologic, and treatment-related factors affecting survival in patients with newly diagnosed diffuse intrinsic pontine glioma (DIPG) treated with radiotherapy.Materials and MethodsPatients aged <30 years who underwent radiotherapy as an initial treatment for DIPG between 2000 and 2018 were included; patients who did not undergo magnetic resonance imaging at diagnosis and those with pathologically diagnosed grade I glioma were excluded. We examined medical records of 162 patients collected from 10 participating centers in Korea. The patients’ clinical, radiological, molecular, and histopathologic characteristics, and treatment responses were evaluated to identify the prognosticators for DIPG and estimate survival outcomes.ResultsThe median follow-up period was 10.8 months (interquartile range, 7.5 to 18.1). The 1- and 2-year overall survival (OS) rates were 53.5% and 19.0%, respectively, with a median OS of 13.1 months. Long-term survival rate (≥ 2 years) was 16.7%, and median OS was 43.6 months. Age (< 10 years), poor performance status, treatment before 2010, and post-radiotherapy necrosis were independently associated with poor OS in multivariate analysis. In patients with increased post-radiotherapy necrosis, the median OS estimates were 13.3 months and 11.4 months with and without bevacizumab, respectively (p=0.138).ConclusionTherapeutic strategy for DIPG has remained unchanged over time, and the associated prognosis remains poor. Our findings suggest that appropriate efforts are needed to reduce the occurrence of post-radiotherapy necrosis. Further well-designed clinical trials are recommended to improve the poor prognosis observed in DIPG patients.
참고문헌 (26)
- Hoffman LM, "Clinical, radiologic, pathologic, and molecular characteristics of long-term survivors of diffuse intrinsic pontine glioma(DIPG) : a collaborative reportfrom the International and European Society for PediatricOncology DIPG Registries", J Clin Oncol, 36, pp.1963~1972 (2018)
- Jackson S, "Clinico-radiologic characteristicsof long-term survivors of diffuse intrinsic pontine glioma", JNeurooncol, 114, pp.339~344 (2013)
- Jansen MH, "Survivalprediction model of children with diffuse intrinsic pontineglioma based on clinical and radiological criteria", NeuroOncol, 17, pp.160~166 (2015)
- Hargrave D, "Diffuse brainstem gliomain children : critical review of clinical trials", Lancet Oncol, 7, pp.241~248 (2006)
- Louis DN, "The 2016 World HealthOrganization classification of tumors of the central nervoussystem : a summary", Acta Neuropathol, 131, pp.803~820 (2016)
- Wu G, "Somatic histone H3 alterations in pediatric diffuseintrinsic pontine gliomas and non-brainstem glioblastomas", Nat Genet, 44, pp.251~253 (2012)
- Langmoen IA, "Management of pediatric pontine gliomas", Childs NervSyst, 7, pp.13~15 (1991)
- Wagner S, "Treatment options in childhood pontine gliomas", J Neurooncol, 79, pp.281~287 (2006)
- Cohen KJ, "Temozolomide in the treatment of children withnewly diagnosed diffuse intrinsic pontine gliomas : a reportfrom the Children’s Oncology Group", Neuro Oncol, 13, pp.410~416 (2011)
- Cooney TM, "Response assessment in diffuse intrinsic pontine glioma : recommendations from the Response Assessment in Pediatric Neuro-Oncology(RAPNO)working group", Lancet Oncol, 21, pp.e330~6 (2020)
- El-Khouly FE, "Diag-nosticsand treatment of diffuse intrinsic pontine glioma : where dowe stand?", J Neurooncol, 145, pp.177~184 (2019)
- Hassan H, "Survival ratesand prognostic predictors of high grade brain stem gliomasin childhood : a systematic review and meta-analysis", J Neurooncol, 135, pp.13~20 (2017)
- Brandel MG, "Survival trends of oligodendroglial tumorpatients and associated clinical practice patterns : a SEER-based analysis", J Neurooncol, 133, pp.173~181 (2017)
- Tejada S, "Biopsy in diffuse pontine gliomas : expertneurosurgeon opinion-a survey from the SIOPE brain tumorgroup", Childs Nerv Syst, 36, pp.705~711 (2020)
- Schwartzentruber J, "Driver mutations in histone H3. 3 and chromatin remodelling genes in paediatric glioblastoma", Nature, 482, pp.226~231 (2012)
- Morales La Madrid A, "Futureclinical trials in DIPG : bringing epigenetics to the clinic", FrontOncol, 5, pp.148~ (2015)
- Tam LT, "MRI-based radiomics for prognosis of pediatric diffuseintrinsic pontine glioma : an international study", NeurooncolAdv, 3, pp.vdab042~ (2021)
- Leach JL, "MR imaging features of diffuse intrinsic pontine gliomaand relationship to overall survival : report from the International DIPG Registry", Neuro Oncol, 22, pp.1647~1657 (2020)
- Szychot E, "Predicting outcome in childhood diffuse midline gliomas using magnetic resonance imaging based texture analysis", J Neuroradiol, 48, pp.243~247 (2021)
- Hummel TR, "A pilot study of bevacizumab-based therapy inpatients with newly diagnosed high-grade gliomas and diffuse intrinsic pontine gliomas", J Neurooncol, 127, pp.53~61 (2016)
- Gururangan S, "Efficacy of bevacizumab plus irinotecan in children with recurrent low-grade gliomas : a Pediatric Brain Tumor Consortium study", Neuro Oncol, 16, pp.310~317 (2014)
- Crotty EE, "Children with DIPG and high-grade glioma treated with temozolomide, irinotecan, and bevacizumab : the Seattle Children’s Hospital experience", J Neurooncol, 148, pp.607~617 (2020)
- Tinkle CL, "Defining optimal target volumes of conformal radiation therapy for diffuse intrinsic pontine glioma", Int J Radiat Oncol Biol Phys, 106, pp.838~847 (2020)
- Broniscer A, "Role of temozolomide after radiotherapy for newly diagnosed diffuse brainstem glioma in children : results of a multiinstitutional study(SJHG-98)", Cancer, 103, pp.133~139 (2005)
- Jalali R, "Prospective evaluation of radiotherapy with concurrent and adjuvant temozolomide in children with newly diagnosed diffuse intrinsic pontine glioma", Int J Radiat Oncol Biol Phys, 77, pp.113~118 (2010)
- Chassot A, "Radiotherapy with concurrent and adjuvant temozolomide in children with newly diagnosed diffuse intrinsic pontine glioma", J Neurooncol, 106, pp.399~407 (2012)
