SHROOM1 is a member of the Apx/Shroom protein family that functions as a regulator of actin cytoskeleton organization and cell morphogenesis. At the molecular level, SHROOM1 contains the characteristic ASD2 domain and links membrane-bound proteins, such as melanoma cell adhesion molecule (MCAM), to the actin cytoskeleton 1. The protein is involved in myofibrillogenesis and sarcomere formation during muscle development, with expression regulated by calcium signaling and epigenetic mechanisms 2. Clinically, SHROOM1 variants have relevance to neural tube defects (NTDs), as the broader SHROOM gene family contributes to neural tube closure morphogenesis 3. Additionally, SHROOM1 hypermethylation has been identified in osteoarthritic cartilage, suggesting epigenetic dysregulation may link this gene to osteoarthritis pathology 4. Notably, SHROOM1 suppression enhances homology-directed DNA repair (HDR) efficiency in human and mouse cells and embryos 5, making SHROOM1 knockdown a potentially useful strategy for improving CRISPR/Cas9-mediated gene editing applications in generating animal models and cell lines for disease research.