Considering the beneficial effects of repopulating microglia in the CNS, it is tempting to hypothesize that replacing microglia in CSF1R-related leukoencephalopathy through a pharmacological depletion and repopulation paradigm may prolong the survival of patients, despite the newly repopulating microglia still carrying the CSF1R gene mutation. Here, CSF1R is linked to Leukoencephalopathy.