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

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The dyslexia susceptibility KIAA0319 gene shows a specific expression pattern during zebrafish development supporting a role beyond neuronal migration. / Gostic, Monika; Martinelli, Angela; Tucker, Carl; Yang, Zhengyi; Gasparoli, Federico Maria; Ewart, Jade-Yi; Dholakia, Kishan; Sillar, Keith; Tello, Javier; Paracchini, Silvia.

In: Journal of Comparative Neurology, Vol. In press, 04.04.2019.

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Gostic, M, Martinelli, A, Tucker, C, Yang, Z, Gasparoli, FM, 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' Journal of Comparative Neurology, vol. In press. https://doi.org/10.1002/cne.24696

APA

Gostic, M., Martinelli, A., Tucker, C., Yang, Z., Gasparoli, F. M., Ewart, J-Y., ... Paracchini, S. (2019). The dyslexia susceptibility KIAA0319 gene shows a specific expression pattern during zebrafish development supporting a role beyond neuronal migration. Journal of Comparative Neurology, In press. https://doi.org/10.1002/cne.24696

Vancouver

Gostic M, Martinelli A, Tucker C, Yang Z, Gasparoli FM, Ewart J-Y et al. The dyslexia susceptibility KIAA0319 gene shows a specific expression pattern during zebrafish development supporting a role beyond neuronal migration. Journal of Comparative Neurology. 2019 Apr 4;In press. https://doi.org/10.1002/cne.24696

Author

Gostic, Monika ; Martinelli, Angela ; Tucker, Carl ; Yang, Zhengyi ; Gasparoli, Federico Maria ; Ewart, Jade-Yi ; Dholakia, Kishan ; Sillar, Keith ; Tello, Javier ; Paracchini, Silvia. / The dyslexia susceptibility KIAA0319 gene shows a specific expression pattern during zebrafish development supporting a role beyond neuronal migration. In: Journal of Comparative Neurology. 2019 ; Vol. In press.

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@article{61a6def7db5f4c44b7714589ee0e1785,
title = "The dyslexia susceptibility KIAA0319 gene shows a specific expression pattern during zebrafish development supporting a role beyond neuronal migration",
abstract = "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 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.",
keywords = "Dyslexia, Development, Zebrafish, Gene expression, Sensory organs, Notochord",
author = "Monika Gostic and Angela Martinelli and Carl Tucker and Zhengyi Yang and Gasparoli, {Federico Maria} and Jade-Yi Ewart and Kishan Dholakia and Keith Sillar and Javier Tello and Silvia Paracchini",
note = "Funding 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: RG160373",
year = "2019",
month = "4",
day = "4",
doi = "10.1002/cne.24696",
language = "English",
volume = "In press",
journal = "Journal of Comparative Neurology",
issn = "0092-7317",

}

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TY - JOUR

T1 - The dyslexia susceptibility KIAA0319 gene shows a specific expression pattern during zebrafish development supporting a role beyond neuronal migration

AU - Gostic, Monika

AU - Martinelli, Angela

AU - Tucker, Carl

AU - Yang, Zhengyi

AU - Gasparoli, Federico Maria

AU - Ewart, Jade-Yi

AU - Dholakia, Kishan

AU - Sillar, Keith

AU - Tello, Javier

AU - Paracchini, Silvia

N1 - Funding 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: RG160373

PY - 2019/4/4

Y1 - 2019/4/4

N2 - 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 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.

AB - 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 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.

KW - Dyslexia

KW - Development

KW - Zebrafish

KW - Gene expression

KW - Sensory organs

KW - Notochord

UR - https://doi.org/10.1101/267617

U2 - 10.1002/cne.24696

DO - 10.1002/cne.24696

M3 - Article

VL - In press

JO - Journal of Comparative Neurology

T2 - Journal of Comparative Neurology

JF - Journal of Comparative Neurology

SN - 0092-7317

ER -

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ID: 252391932