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Ash1l  potentially contributes to neurodevelopmental diseases. Although specific  Ash1l  mutations are rare, they have led to informative studies in animal models that may bring therapeutic advances.  Ash1l  is highly expressed in the brain and correlates with the neuropathology of Tourette syndrome (TS), autism spectrum disorder, and intellectual disability during development, implicating shared epigenetic factors and overlapping neuropathological mechanisms. Functional convergence of  Ash1l  generated several significant signaling pathways: chromatin remodeling and transcriptional regulation, protein synthesis and cellular metabolism, and synapse development and function. Here, we systematically review the literature on  Ash1l , including its discovery, expression, function, regulation, implication in the nervous system, signaling pathway, mutations, and putative involvement in TS and other neurodevelopmental traits. Such findings highlight  Ash1l  pleiotropy and the necessity of transcending a single gene to complicated mechanisms of network convergence underlying these diseases. With the progress in functional genomic analysis (highlighted in this review), and although the importance and necessity of  Ash1l  becomes increasingly apparent in the medical field, further research is required to discover the precise function and molecular regulatory mechanisms related to  Ash1l . Thus, a new perspective is proposed for basic scientific research and clinical interventions for cross‐disorder diseases.

Role of Ash1l in Tourette syndrome and other neurodevelopmental disorders