Medulloblastoma is an extremely rare disease in adults. After surgery, the mainstay of treatment is based on chemotherapy and radiotherapy, that provide 5 year suvival rates of 50-80% according to risk factors. Post-therapy nauronal maturation in medulloblastoma is a rare event that has been described only in pediatric age. Here we report the case of neuronal maturation after chemotherapy and radiotherapy in an adult patient. We also assessed that molecular SHH grouping with Illumina Human Methylation 450 k Bead Chip arrays showing that this molecular feature was stable during this maturation. We also discuss about treatment approaches in this rare situation.