biostudies-literatureWang YNational Institute of Arthritis and Musculoskeletal and Skin DiseasesNational Natural Science Foundation of ChinaChina Postdoctoral Science FoundationNational Institutes of HealthNIAMS NIH HHS82-90https://www.ebi.ac.uk/biostudies/studies/S-EPMC8792996biostudies-literatureUnknown482Adolescent idiopathic scoliosis (AIS) is a common pediatric musculoskeletal disorder worldwide, characterized by atypical spine curvatures in otherwise healthy children. Human genetic studies have identified candidate genes associated with AIS, however, only a few of these have been shown to recapitulate adult-viable scoliosis in animal models. Using an F0 CRISPR screening approach in zebrafish, we demonstrate that disruption of the dynein axonemal heavy chain 10 (dnah10) gene results in recessive adult-viable scoliosis in zebrafish. Using a stably segregating dnah10 mutant zebrafish, we showed that the ependymal monocilia lining the hindbrain and spinal canal displayed reduced beat frequency, which was correlated with the disassembly of the Reissner fiber and the onset of body curvatures.Developmental biologyThe axonemal dynein heavy chain 10 gene is essential for monocilia motility and spine alignment in zebrafish.PMC879299681472145817722982021M70364082072390R01 AR072009AR072009Troutwine BRZhang HWang YGray RSfalseThe axonemal dynein heavy chain 10 gene is essential for monocilia motility and spine alignment in zebrafish.Adolescent idiopathic scoliosis (AIS) is a common pediatric musculoskeletal disorder worldwide, characterized by atypical spine curvatures in otherwise healthy children. Human genetic studies have identified candidate genes associated with AIS, however, only a few of these have been shown to recapitulate adult-viable scoliosis in animal models. Using an F0 CRISPR screening approach in zebrafish, we demonstrate that disruption of the dynein axonemal heavy chain 10 (dnah10) gene results in recessive adult-viable scoliosis in zebrafish. Using a stably segregating dnah10 mutant zebrafish, we showed that the ependymal monocilia lining the hindbrain and spinal canal displayed reduced beat frequency, which was correlated with the disassembly of the Reissner fiber and the onset of body curvatures.2022-01-01T00:00:00Z2022 Feb2025-04-19T17:56:56.435Z2025-04-19T17:56:56.435ZS-EPMC87929963491502210.1016/j.ydbio.2021.12.001