Furthermore, it remains to be elucidated whether levels of CSF1 and IL‐34 in the plasma and cerebrospinal fluid as well as ex vivo CSF1R functional studies of patient‐derived monocytes might serve to investigate a potential pharmacological benefit of CSF1R inhibition in patients with HDLS. Here, CSF1R is linked to Hereditary diffuse leukoencephalopathy with axonal spheroids and pigmented glia.