The report on a detection of a de novo 1.1-Mbp duplication, involving a genomic region harboring three genes among which PTPRM, in a patient with hemiplegic cerebral palsy (Zarrei et al., 2018) actually comes close to the arterial phenotype noted in Ptprm knockout mice because this disease is characterized by one-sided defective posture and movement that is due to a vascular insult, a venous infarction, or brain malformation. Here, PTPRM is linked to cerebral malformation.