Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.

Axon pathfinding is a subfield of neural development by which neurons send out axons to reach the correct targets. In particular, motoneurons extend their axons toward skeletal muscles, leading to spontaneous motor activity. In this study, we identified the zebrafish Ccdc80 and Ccdc80-like1 (Ccdc80-...

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Autores principales:	Chiara Brusegan, Anna Pistocchi, Andrea Frassine, Isabella Della Noce, Filippo Schepis, Franco Cotelli
Formato:	article
Lenguaje:	EN
Publicado:	Public Library of Science (PLoS) 2012
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/76622cf864b84eeea23a4ec8a680891d
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spelling	oai:doaj.org-article:76622cf864b84eeea23a4ec8a680891d2021-11-18T07:27:05ZCcdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.1932-620310.1371/journal.pone.0031851https://doaj.org/article/76622cf864b84eeea23a4ec8a680891d2012-01-01T00:00:00Zhttps://www.ncbi.nlm.nih.gov/pmc/articles/pmid/22384085/pdf/?tool=EBIhttps://doaj.org/toc/1932-6203Axon pathfinding is a subfield of neural development by which neurons send out axons to reach the correct targets. In particular, motoneurons extend their axons toward skeletal muscles, leading to spontaneous motor activity. In this study, we identified the zebrafish Ccdc80 and Ccdc80-like1 (Ccdc80-l1) proteins in silico on the basis of their high aminoacidic sequence identity with the human CCDC80 (Coiled-Coil Domain Containing 80). We focused on ccdc80-l1 gene that is expressed in nervous and non-nervous tissues, in particular in territories correlated with axonal migration, such as adaxial cells and muscle pioneers. Loss of ccdc80-l1 in zebrafish embryos induced motility issues, although somitogenesis and myogenesis were not impaired. Our results strongly suggest that ccdc80-l1 is involved in axon guidance of primary and secondary motoneurons populations, but not in their proper formation. ccdc80-l1 has a differential role as regards the development of ventral and dorsal motoneurons, and this is consistent with the asymmetric distribution of the transcript. The axonal migration defects observed in ccdc80-l1 loss-of-function embryos are similar to the phenotype of several mutants with altered Hedgehog activity. Indeed, we reported that ccdc80-l1 expression is positively regulated by the Hedgehog pathway in adaxial cells and muscle pioneers. These findings strongly indicate ccdc80-l1 as a down-stream effector of the Hedgehog pathway.Chiara BruseganAnna PistocchiAndrea FrassineIsabella Della NoceFilippo SchepisFranco CotelliPublic Library of Science (PLoS)articleMedicineRScienceQENPLoS ONE, Vol 7, Iss 2, p e31851 (2012)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Chiara Brusegan Anna Pistocchi Andrea Frassine Isabella Della Noce Filippo Schepis Franco Cotelli Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.
description	Axon pathfinding is a subfield of neural development by which neurons send out axons to reach the correct targets. In particular, motoneurons extend their axons toward skeletal muscles, leading to spontaneous motor activity. In this study, we identified the zebrafish Ccdc80 and Ccdc80-like1 (Ccdc80-l1) proteins in silico on the basis of their high aminoacidic sequence identity with the human CCDC80 (Coiled-Coil Domain Containing 80). We focused on ccdc80-l1 gene that is expressed in nervous and non-nervous tissues, in particular in territories correlated with axonal migration, such as adaxial cells and muscle pioneers. Loss of ccdc80-l1 in zebrafish embryos induced motility issues, although somitogenesis and myogenesis were not impaired. Our results strongly suggest that ccdc80-l1 is involved in axon guidance of primary and secondary motoneurons populations, but not in their proper formation. ccdc80-l1 has a differential role as regards the development of ventral and dorsal motoneurons, and this is consistent with the asymmetric distribution of the transcript. The axonal migration defects observed in ccdc80-l1 loss-of-function embryos are similar to the phenotype of several mutants with altered Hedgehog activity. Indeed, we reported that ccdc80-l1 expression is positively regulated by the Hedgehog pathway in adaxial cells and muscle pioneers. These findings strongly indicate ccdc80-l1 as a down-stream effector of the Hedgehog pathway.
format	article
author	Chiara Brusegan Anna Pistocchi Andrea Frassine Isabella Della Noce Filippo Schepis Franco Cotelli
author_facet	Chiara Brusegan Anna Pistocchi Andrea Frassine Isabella Della Noce Filippo Schepis Franco Cotelli
author_sort	Chiara Brusegan
title	Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.
title_short	Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.
title_full	Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.
title_fullStr	Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.
title_full_unstemmed	Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.
title_sort	ccdc80-l1 is involved in axon pathfinding of zebrafish motoneurons.
publisher	Public Library of Science (PLoS)
publishDate	2012
url	https://doaj.org/article/76622cf864b84eeea23a4ec8a680891d
work_keys_str_mv	AT chiarabrusegan ccdc80l1isinvolvedinaxonpathfindingofzebrafishmotoneurons AT annapistocchi ccdc80l1isinvolvedinaxonpathfindingofzebrafishmotoneurons AT andreafrassine ccdc80l1isinvolvedinaxonpathfindingofzebrafishmotoneurons AT isabelladellanoce ccdc80l1isinvolvedinaxonpathfindingofzebrafishmotoneurons AT filipposchepis ccdc80l1isinvolvedinaxonpathfindingofzebrafishmotoneurons AT francocotelli ccdc80l1isinvolvedinaxonpathfindingofzebrafishmotoneurons
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Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.

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