Here, we show that the addition of healthy microglia reduces both neuropathology and symptom presentation in a zebrafish model of RNASET2-deficient leukodystrophy—a disorder which sits at the intersection of leukodystrophies, lysosomal storage diseases, and a family of autoinflammatory disorders characterized by upregulation of the interferon response, known as interferonopathies (26, –28). This evidence concerns the gene RNASET2 and leukodystrophy.