Jian Peng1, Hao Zhou2, Oliver Tang3, Ken Chang4, Panpan Wang1, Xiaowei Zeng1, Qin Shen5, Jing Wu5, Yanhe Xiao1, Sohil H Patel6, Chongyu Hu7, Ke Jin8, Bo Xiao2, Jerrold Boxerman9, Xiaoping Gao7, Patrick Y Wen10, Harrison X Bai9,11, Raymond Y Huang12, Li Yang1. 1. Department of Neurology, Second Xiangya Hospital of Central South University, Changsha, Hunan, China. 2. Department of Neurology, Xiangya Hospital of Central South University, Changsha, Hunan, China. 3. Warren Alpert Medical School of Brown University, Providence, Rhode Island, USA. 4. Department of Radiology, Athinoula A. Martinos Center for Biomedical Imaging, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts, USA. 5. Department of Radiology, Second Xiangya Hospital of Central South University, Changsha, Hunan, China. 6. Department of Radiology and Medical Imaging, University of Virginia Health System, Charlottesville, Virginia, USA. 7. Department of Neurology, Hunan Provincial People's Hospital, Changsha, Hunan, China. 8. Department of Radiology, Hunan Children's Hospital, Changsha, Hunan, China. 9. Department of Diagnostic Imaging, Rhode Island Hospital and Alpert Medical School of Brown University, Providence, Rhode Island, USA. 10. Center for Neuro-Oncology, Dana Farber Cancer Institute, Boston, Massachusetts, USA. 11. Department of Radiology, Xiangya Hospital of Central South University, Changsha, Hunan, China. 12. Department of Radiology, Brigham and Women's Hospital, Boston, Massachusetts, USA.
Abstract
BACKGROUND: Although the Response Assessment in Pediatric Neuro-Oncology (RAPNO) working group has made recommendations for response assessment in patients with medulloblastoma (MBL) and leptomeningeal seeding tumors, these criteria have yet to be evaluated. METHODS: We examined MR imaging and clinical data in a multicenter retrospective cohort of 269 patients with MBL diagnoses, high grade glioma, embryonal tumor, germ cell tumor, or choroid plexus papilloma. Interobserver agreement, objective response (OR) rates, and progression-free survival (PFS) were calculated. Landmark analyses were performed for OR and progression status at 0.5, 1.0, and 1.5 years after treatment initiation. Cox proportional hazards models were used to determine the associations between OR and progression with overall survival (OS). Subgroup analyses based on tumor subgroup and treatment modality were performed. RESULTS: The median follow-up time was 4.0 years. In all patients, the OR rate was .0.565 (95% CI: 0.505-0.625) by RAPNO. The interobserver agreement of OR determination between 2 raters (a neuroradiologist and a neuro-oncologist) for the RAPNO criteria in all patients was 83.8% (k statistic = 0.815; P < 0.001). At 0.5-, 1.0-, and 1.5-year landmarks, both OR status and PFS determined by RAPNO were predictive of OS (hazard ratios [HRs] for 1-year landmark: OR HR = 0.079, P < 0.001; PFS HR = 10.192, P < 0.001). In subgroup analysis, OR status and PFS were predictive of OS for all tumor subtypes and treatment modalities. CONCLUSION: RAPNO criteria showed excellent consistency in the treatment response evaluation of MBL and other leptomeningeal seeding tumors. OR and PFS determined by RAPNO criteria correlated with OS.
BACKGROUND: Although the Response Assessment in Pediatric Neuro-Oncology (RAPNO) working group has made recommendations for response assessment in patients with medulloblastoma (MBL) and leptomeningeal seeding tumors, these criteria have yet to be evaluated. METHODS: We examined MR imaging and clinical data in a multicenter retrospective cohort of 269 patients with MBL diagnoses, high grade glioma, embryonal tumor, germ cell tumor, or choroid plexus papilloma. Interobserver agreement, objective response (OR) rates, and progression-free survival (PFS) were calculated. Landmark analyses were performed for OR and progression status at 0.5, 1.0, and 1.5 years after treatment initiation. Cox proportional hazards models were used to determine the associations between OR and progression with overall survival (OS). Subgroup analyses based on tumor subgroup and treatment modality were performed. RESULTS: The median follow-up time was 4.0 years. In all patients, the OR rate was .0.565 (95% CI: 0.505-0.625) by RAPNO. The interobserver agreement of OR determination between 2 raters (a neuroradiologist and a neuro-oncologist) for the RAPNO criteria in all patients was 83.8% (k statistic = 0.815; P < 0.001). At 0.5-, 1.0-, and 1.5-year landmarks, both OR status and PFS determined by RAPNO were predictive of OS (hazard ratios [HRs] for 1-year landmark: OR HR = 0.079, P < 0.001; PFS HR = 10.192, P < 0.001). In subgroup analysis, OR status and PFS were predictive of OS for all tumor subtypes and treatment modalities. CONCLUSION: RAPNO criteria showed excellent consistency in the treatment response evaluation of MBL and other leptomeningeal seeding tumors. OR and PFS determined by RAPNO criteria correlated with OS.
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