Bicra, also known as GLTSCR1, encodes a subunit of the non-canonical BAF (ncBAF) chromatin remodeling complex. As a member of the SWI/SNF family, it is involved in chromatin remodeling, which is crucial for regulating gene expression, and thus has overall biological importance in various biological processes [1].

In zebrafish, a mutation of bicra mimicking a loss-of-function variant leads to craniofacial defects, similar to the dysmorphic facial features seen in humans with pathogenic BICRA variants [1]. In Drosophila, loss of Bicra impairs learning and choice abilities, specifically in male courtship learning and choice performance, indicating its role in cognition-related behaviors [2]. In mice, homozygous loss of Bicra using CRISPR/Cas9 results in blood-related defects, including defective erythroblastic islands and irregularly sized red blood cells due to a defect in fetal liver macrophages responsible for erythrocyte maturation [5].

In conclusion, Bicra plays essential roles in multiple biological processes, as revealed through studies in zebrafish, Drosophila, and mouse models. These model-based research findings show its significance in craniofacial development, cognitive behaviors, and erythrocyte maturation. The loss-of-function studies in these models contribute to understanding the role of Bicra in related human diseases, such as Coffin-Siris syndrome 12, which is associated with diverse neurodevelopmental delays [1,3,4].