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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Public Library of Science (PLoS)
2012-01-01
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| Series: | PLoS ONE |
| Online Access: | https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0031851&type=printable |
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| 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 |
| collection | DOAJ |
| 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 |
| id | doaj-art-76622cf864b84eeea23a4ec8a680891d |
| institution | DOAJ |
| issn | 1932-6203 |
| language | English |
| publishDate | 2012-01-01 |
| publisher | Public Library of Science (PLoS) |
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| series | PLoS ONE |
| spelling | doaj-art-76622cf864b84eeea23a4ec8a680891d2025-08-20T02:43:54ZengPublic Library of Science (PLoS)PLoS ONE1932-62032012-01-0172e3185110.1371/journal.pone.0031851Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons.Chiara BruseganAnna PistocchiAndrea FrassineIsabella Della NoceFilippo SchepisFranco CotelliAxon 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.https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0031851&type=printable |
| spellingShingle | Chiara Brusegan Anna Pistocchi Andrea Frassine Isabella Della Noce Filippo Schepis Franco Cotelli Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons. PLoS ONE |
| title | 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_short | Ccdc80-l1 Is involved in axon pathfinding of zebrafish motoneurons. |
| title_sort | ccdc80 l1 is involved in axon pathfinding of zebrafish motoneurons |
| url | https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0031851&type=printable |
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