Abstract
DNA methylation plays an important role in the regulation of gene expression. Its deficits in the brain cause various neurological diseases, including autism, schizophrenia and mood disorders. The zebrafish (Danio rerio) is a promising model organism in biomedicine. Given its high genetic and physiological homology with humans, studying genome methylation deficits in zebrafish can help elucidate the molecular processes underlying the etiology and pathogenesis of various neurological diseases, as well as develop novel therapies. Here, we discuss the mechanisms of DNA methylation in the brain and the diseases associated with its dysregulation in humans, as well as their genetic and pharmacological models in zebrafish. We also evaluate the limitations of zebrafish models and possible directions for further research in this field. Mounting evidence summarized here supports zebrafish as an effective model for elucidating the molecular mechanisms of brain pathologies associated with compromised DNA methylation.