Patients’ treatment and molecular alteration. Swimmer plot representing patients’ outcome starting at the beginning of the first SMOi treatment (a). Patients are segregated by SHH activation downstream SMO, above the dotted line, or SHH activation upstream SMO, below the dotted line. The first column described initial SHH pathway molecular alteration based on CGH, methylome analysis, and/or sequencing. Event, including responses, progressive disease, discontinuation, or death are represented on the patient line as well as other relevant treatment targeting SHH pathway or resistance. For patients who underwent a new biopsy at progression, SMO mutation are reported with their allelic fraction in the tumor biopsy. Unsupervised clustering of methylation pattern according to medulloblastoma subgroups using UMAP showed that the 4 patients were clustering with the previously published SHH medulloblastoma (b). SHH subgroups affiliations between infant (INF) or child and adolescent (CHL AD) of patients tumors were consistent with age at diagnosis. Patient #6 was 13 years old while the remaining 3 patients were of 4 years old or below at diagnosis (c). copy number profils infered from methylation array showing recurrent copy number alteration in SHH medulloblastoma. SHH, Sonic Hedgehog; SMOi, smoothened inhibitor.