ArticleScientific reports2022
KIAA0319 influences cilia length, cell migration and mechanical cell-substrate interaction.
Article in Scientific reports, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers.
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Who cites it
9 citing papers in PubMed, 12 citations in OpenAlex.
- Genetic Variants Linked to Dyslexia Co-Morbid ADHD: A Case Study of a Pakistani Outpatient.Journal of population therapeutics and clinical pharmacology = Journal de la therapeutique des populations et de la pharmacologie clinique · 2024Article
- Developmental dyslexia susceptibility genes DNAAF4, DCDC2, and NRSN1 are associated with brain function in fluently reading adolescents and young adults.Cerebral cortex (New York, N.Y. : 1991) · 2024Article
- Single-cell RNA-seq data analysis reveals functionally relevant biomarkers of early brain development and their regulatory footprints in human embryonic stem cells (hESCs).Briefings in bioinformatics · 2024Article
- Genetic Modifications of Developmental Dyslexia and Its Representation Using In Vivo, In Vitro Model.Global medical genetics · 2024Review
- Specific Learning Disorders: Variation Analysis of 15 Candidate Genes in 9 Multiplex Families.Medicina (Kaunas, Lithuania) · 2023Article
- Dyslexia-Related Hearing Loss Occurs Mainly through the Abnormal Spontaneous Electrical Activity of Spiral Ganglion Neurons.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2023Article
- Macular Anatomy Differs in Dyslexic Subjects.Journal of clinical medicine · 2023Article
- Animal models of developmental dyslexia.Frontiers in neuroscience · 2022Review
- Insights into Dyslexia Genetics Research from the Last Two Decades.Brain sciences · 2021Review
Corrections and comments
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Authors and funding
7 authors at 1 institution in 1 country.
Funding
Abstract
Following its association with dyslexia in multiple genetic studies, the KIAA0319 gene has been extensively investigated in different animal models but its function in neurodevelopment remains poorly understood. We developed the first human cellular knockout model for KIAA0319 in RPE1 retinal pigment epithelia cells via CRISPR-Cas9n to investigate its role in processes suggested but not confirmed in previous studies, including cilia formation and cell migration. We observed in the KIAA0319 knockout increased cilia length and accelerated cell migration. Using Elastic Resonator Interference Stress Microscopy (ERISM), we detected an increase in cellular force for the knockout cells that was restored by a rescue experiment. Combining ERISM and immunostaining we show that RPE1 cells exert highly dynamic, piconewton vertical pushing forces through actin-rich protrusions that are surrounded by vinculin-rich pulling sites. This protein arrangement and force pattern has previously been associated to podosomes in other cells. KIAA0319 depletion reduces the fraction of cells forming these actin-rich protrusions. Our results suggest an involvement of KIAA0319 in cilia biology and cell-substrate force regulation.
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