The dyslexia susceptibility KIAA0319 gene shows a specific expression pattern during zebrafish development supporting a role beyond neuronal migration.

Autor: Gostic M; School of Medicine, University of St Andrews, St Andrews, UK.; Biomedical Sciences Research Complex, University of St Andrews, St Andrews, UK., Martinelli A; School of Medicine, University of St Andrews, St Andrews, UK.; Biomedical Sciences Research Complex, University of St Andrews, St Andrews, UK., Tucker C; College of Medicine and Veterinary Medicine, The University of Edinburgh, Edinburgh, UK., Yang Z; SUPA, School of Physics and Astronomy, University of St Andrews, St Andrews, UK.; School of Psychology and Neuroscience, University of St Andrews, St Andrews, UK., Gasparoli F; School of Medicine, University of St Andrews, St Andrews, UK., Ewart JY; School of Medicine, University of St Andrews, St Andrews, UK.; School of Psychology and Neuroscience, University of St Andrews, St Andrews, UK., Dholakia K; Biomedical Sciences Research Complex, University of St Andrews, St Andrews, UK.; SUPA, School of Physics and Astronomy, University of St Andrews, St Andrews, UK., Sillar KT; School of Psychology and Neuroscience, University of St Andrews, St Andrews, UK., Tello JA; School of Medicine, University of St Andrews, St Andrews, UK.; Biomedical Sciences Research Complex, University of St Andrews, St Andrews, UK., Paracchini S; School of Medicine, University of St Andrews, St Andrews, UK.; Biomedical Sciences Research Complex, University of St Andrews, St Andrews, UK.
Jazyk: angličtina
Zdroj: The Journal of comparative neurology [J Comp Neurol] 2019 Nov 01; Vol. 527 (16), pp. 2634-2643. Date of Electronic Publication: 2019 Apr 16.
DOI: 10.1002/cne.24696
Abstrakt: Dyslexia is a common neurodevelopmental disorder caused by a significant genetic component. The KIAA0319 gene is one of the most robust dyslexia susceptibility factors but its function remains poorly understood. Initial RNA-interference studies in rats suggested a role in neuronal migration whereas subsequent work with double knock-out mouse models for both Kiaa0319 and its paralogue Kiaa0319-like reported effects in the auditory system but not in neuronal migration. To further understand the role of KIAA0319 during neurodevelopment, we carried out an expression study of its zebrafish orthologue at different embryonic stages. We used different approaches including RNAscope in situ hybridization combined with light-sheet microscopy. The results show particularly high expression during the first few hours of development. Later, expression becomes localized in well-defined structures. In addition to high expression in the brain, we report for the first time expression in the eyes and the notochord. Surprisingly, kiaa0319-like, which generally shows a similar expression pattern to kiaa0319, was not expressed in the notochord suggesting a distinct role for kiaa0319 in this structure. This observation was supported by the identification of notochord enhancers enriched upstream of the KIAA0319 transcription start site, in both zebrafish and humans. This study supports a developmental role for KIAA0319 in the brain as well as in other developing structures, particularly in the notochord which, is key for establishing body patterning in vertebrates.
(© 2019 The Authors. The Journal of Comparative Neurology published by Wiley Periodicals, Inc.)
Databáze: MEDLINE
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