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dc.contributor.authorGostic, Monika
dc.contributor.authorMartinelli, Angela
dc.contributor.authorTucker, Carl
dc.contributor.authorYang, Zhengyi
dc.contributor.authorGasparoli, Federico Maria
dc.contributor.authorEwart, Jade-Yi
dc.contributor.authorDholakia, Kishan
dc.contributor.authorSillar, Keith
dc.contributor.authorTello, Javier
dc.contributor.authorParacchini, Silvia
dc.identifier.citationGostic , M , Martinelli , A , Tucker , C , Yang , Z , Gasparoli , F M , Ewart , J-Y , Dholakia , K , Sillar , K , Tello , J & Paracchini , S 2019 , ' The dyslexia susceptibility KIAA0319 gene shows a specific expression pattern during zebrafish development supporting a role beyond neuronal migration ' , The Journal of Comparative Neurology , vol. 527 , no. 16 , pp. 2634-2643 .
dc.identifier.otherPURE: 252391932
dc.identifier.otherPURE UUID: 61a6def7-db5f-4c44-b771-4589ee0e1785
dc.identifier.otherWOS: 000483648200002
dc.identifier.otherScopus: 85064500894
dc.identifier.otherORCID: /0000-0001-9934-8602/work/60428094
dc.identifier.otherORCID: /0000-0001-6637-2155/work/64034510
dc.identifier.otherORCID: /0000-0003-0171-3814/work/64393778
dc.identifier.otherWOS: 000483648200002
dc.descriptionFunding information: Carnegie Trust for the Universities of Scotland, Grant/Award Number: 50341; Engineering and Physical Sciences Research Council, Grant/Award Numbers: EP/R004854/1, EP/P030017/1; EuFishBioMed; Northwood Trust; Royal Society, Grant/Award Number: RG160373en
dc.description.abstractDyslexia 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 localised 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.
dc.relation.ispartofThe Journal of Comparative Neurologyen
dc.rights© 2019 The Authors. The Journal of Comparative Neurology published by Wiley Periodicals, Inc. This is an open access article under the terms of the Creative Commons Attribution License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.en
dc.subjectGene expressionen
dc.subjectSensory organsen
dc.subjectBF Psychologyen
dc.subjectQH301 Biologyen
dc.subjectQH426 Geneticsen
dc.subjectRC0321 Neuroscience. Biological psychiatry. Neuropsychiatryen
dc.titleThe dyslexia susceptibility KIAA0319 gene shows a specific expression pattern during zebrafish development supporting a role beyond neuronal migrationen
dc.typeJournal articleen
dc.contributor.sponsorThe Royal Societyen
dc.contributor.sponsorCarnegie Trusten
dc.description.versionPublisher PDFen
dc.contributor.institutionUniversity of St Andrews. School of Medicineen
dc.contributor.institutionUniversity of St Andrews. Cellular Medicine Divisionen
dc.contributor.institutionUniversity of St Andrews. School of Physics and Astronomyen
dc.contributor.institutionUniversity of St Andrews. School of Psychology and Neuroscienceen
dc.contributor.institutionUniversity of St Andrews. Institute of Behavioural and Neural Sciencesen
dc.contributor.institutionUniversity of St Andrews. Biomedical Sciences Research Complexen
dc.contributor.institutionUniversity of St Andrews. Centre for Biophotonicsen
dc.description.statusPeer revieweden

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