Drosophila CASK regulates brain size and neuronal morphogenesis, providing a genetic model of postnatal microcephaly suitable for drug discovery.
Judith A TelloLinan JiangYitshak ZoharLinda L RestifoPublished in: Neural development (2023)
These biological and engineering advances set the stage for drug discovery using the Drosophila model of CASK-related disorders. The bushy phenotype provides a cell-based assay for compound screening. Nearly a dozen genes encoding CASK-binding proteins or transcriptional targets also have brain-development mutant phenotypes, including ID. Hence, drugs that improve CASK phenotypes might also benefit children with disorders due to mutant CASK partners.
Keyphrases
- drug discovery
- resting state
- cerebral ischemia
- white matter
- genome wide
- zika virus
- young adults
- single cell
- functional connectivity
- gene expression
- preterm infants
- high throughput
- transcription factor
- wild type
- cell therapy
- intellectual disability
- dna methylation
- blood brain barrier
- brain injury
- drug induced
- mesenchymal stem cells
- heat shock
- antiretroviral therapy