Cargando…
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...
Autores principales: | , , , , , , , , , , , , , |
---|---|
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 |
_version_ | 1782206282421239808 |
---|---|
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. |
format | Online Article Text |
id | pubmed-3116825 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2011 |
publisher | Public Library of Science |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT massinensatu increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT hokkanenmarieestelle increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT matssonhans increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT tammimieskristiina increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT tapiapaezisabel increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT dahlstromheuservanina increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT kujapanulajuha increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT burghoornjan increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT jeppssonkristiane increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT swobodapeter increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT peyrardjanvidmyriam increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT toftgardrune increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT castreneero increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons AT kerejuha increasedexpressionofthedyslexiacandidategenedcdc2affectslengthandsignalingofprimaryciliainneurons |