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Increased Expression of the Dyslexia Candidate Gene DCDC2 Affects Length and Signaling of Primary Cilia in Neurons

DCDC2 is one of the candidate susceptibility genes for dyslexia. It belongs to the superfamily of doublecortin domain containing proteins that bind to microtubules, and it has been shown to be involved in neuronal migration. We show that the Dcdc2 protein localizes to the primary cilium in primary r...

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Autores principales: Massinen, Satu, Hokkanen, Marie-Estelle, Matsson, Hans, Tammimies, Kristiina, Tapia-Páez, Isabel, Dahlström-Heuser, Vanina, Kuja-Panula, Juha, Burghoorn, Jan, Jeppsson, Kristian E., Swoboda, Peter, Peyrard-Janvid, Myriam, Toftgård, Rune, Castrén, Eero, Kere, Juha
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Public Library of Science 2011
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3116825/
https://www.ncbi.nlm.nih.gov/pubmed/21698230
http://dx.doi.org/10.1371/journal.pone.0020580
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author Massinen, Satu
Hokkanen, Marie-Estelle
Matsson, Hans
Tammimies, Kristiina
Tapia-Páez, Isabel
Dahlström-Heuser, Vanina
Kuja-Panula, Juha
Burghoorn, Jan
Jeppsson, Kristian E.
Swoboda, Peter
Peyrard-Janvid, Myriam
Toftgård, Rune
Castrén, Eero
Kere, Juha
author_facet Massinen, Satu
Hokkanen, Marie-Estelle
Matsson, Hans
Tammimies, Kristiina
Tapia-Páez, Isabel
Dahlström-Heuser, Vanina
Kuja-Panula, Juha
Burghoorn, Jan
Jeppsson, Kristian E.
Swoboda, Peter
Peyrard-Janvid, Myriam
Toftgård, Rune
Castrén, Eero
Kere, Juha
author_sort Massinen, Satu
collection PubMed
description DCDC2 is one of the candidate susceptibility genes for dyslexia. It belongs to the superfamily of doublecortin domain containing proteins that bind to microtubules, and it has been shown to be involved in neuronal migration. We show that the Dcdc2 protein localizes to the primary cilium in primary rat hippocampal neurons and that it can be found within close proximity to the ciliary kinesin-2 subunit Kif3a. Overexpression of DCDC2 increases ciliary length and activates Shh signaling, whereas downregulation of Dcdc2 expression enhances Wnt signaling, consistent with a functional role in ciliary signaling. Moreover, DCDC2 overexpression in C. elegans causes an abnormal neuronal phenotype that can only be seen in ciliated neurons. Together our results suggest a potential role for DCDC2 in the structure and function of primary cilia.
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spelling pubmed-31168252011-06-22 Increased Expression of the Dyslexia Candidate Gene DCDC2 Affects Length and Signaling of Primary Cilia in Neurons Massinen, Satu Hokkanen, Marie-Estelle Matsson, Hans Tammimies, Kristiina Tapia-Páez, Isabel Dahlström-Heuser, Vanina Kuja-Panula, Juha Burghoorn, Jan Jeppsson, Kristian E. Swoboda, Peter Peyrard-Janvid, Myriam Toftgård, Rune Castrén, Eero Kere, Juha PLoS One Research Article DCDC2 is one of the candidate susceptibility genes for dyslexia. It belongs to the superfamily of doublecortin domain containing proteins that bind to microtubules, and it has been shown to be involved in neuronal migration. We show that the Dcdc2 protein localizes to the primary cilium in primary rat hippocampal neurons and that it can be found within close proximity to the ciliary kinesin-2 subunit Kif3a. Overexpression of DCDC2 increases ciliary length and activates Shh signaling, whereas downregulation of Dcdc2 expression enhances Wnt signaling, consistent with a functional role in ciliary signaling. Moreover, DCDC2 overexpression in C. elegans causes an abnormal neuronal phenotype that can only be seen in ciliated neurons. Together our results suggest a potential role for DCDC2 in the structure and function of primary cilia. Public Library of Science 2011-06-16 /pmc/articles/PMC3116825/ /pubmed/21698230 http://dx.doi.org/10.1371/journal.pone.0020580 Text en Massinen et al. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are properly credited.
spellingShingle Research Article
Massinen, Satu
Hokkanen, Marie-Estelle
Matsson, Hans
Tammimies, Kristiina
Tapia-Páez, Isabel
Dahlström-Heuser, Vanina
Kuja-Panula, Juha
Burghoorn, Jan
Jeppsson, Kristian E.
Swoboda, Peter
Peyrard-Janvid, Myriam
Toftgård, Rune
Castrén, Eero
Kere, Juha
Increased Expression of the Dyslexia Candidate Gene DCDC2 Affects Length and Signaling of Primary Cilia in Neurons
title Increased Expression of the Dyslexia Candidate Gene DCDC2 Affects Length and Signaling of Primary Cilia in Neurons
title_full Increased Expression of the Dyslexia Candidate Gene DCDC2 Affects Length and Signaling of Primary Cilia in Neurons
title_fullStr Increased Expression of the Dyslexia Candidate Gene DCDC2 Affects Length and Signaling of Primary Cilia in Neurons
title_full_unstemmed Increased Expression of the Dyslexia Candidate Gene DCDC2 Affects Length and Signaling of Primary Cilia in Neurons
title_short Increased Expression of the Dyslexia Candidate Gene DCDC2 Affects Length and Signaling of Primary Cilia in Neurons
title_sort increased expression of the dyslexia candidate gene dcdc2 affects length and signaling of primary cilia in neurons
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3116825/
https://www.ncbi.nlm.nih.gov/pubmed/21698230
http://dx.doi.org/10.1371/journal.pone.0020580
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