2012
DOI: 10.1371/journal.pone.0031851
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ccdc80-l1 Is Involved in Axon Pathfinding of Zebrafish Motoneurons

Abstract: 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 tis… Show more

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Cited by 13 publications
(13 citation statements)
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“…D and 2C), has been implicated in adipogenesis in mice [Tremblay et al, ]; hence, one could hypothesize that CCDC80 could also be responsible for the mirror phenotype in the abnormal postnatal growth observed in patients with microdeletions (overgrowth) [Molin et al, ; Shuvarikov et al, ] and microduplications (undergrowth) [Karavitakis et al, ] at chromosome 3q13.2–q13.31. Furthermore, loss of the homologous gene ccdc80‐l1 induces motility issues in zebrafish [Brusegan et al, ]; so that, Karavitakis et al [] have suggested its potential implication in the muscular hypotonia seen in patients with dose alterations at 3q13.2–q13.31 [Karavitakis et al, ], a phenotypic manifestation observed in our patient who also has only one copy of CCDC80 .…”
Section: Discussionmentioning
confidence: 69%
“…D and 2C), has been implicated in adipogenesis in mice [Tremblay et al, ]; hence, one could hypothesize that CCDC80 could also be responsible for the mirror phenotype in the abnormal postnatal growth observed in patients with microdeletions (overgrowth) [Molin et al, ; Shuvarikov et al, ] and microduplications (undergrowth) [Karavitakis et al, ] at chromosome 3q13.2–q13.31. Furthermore, loss of the homologous gene ccdc80‐l1 induces motility issues in zebrafish [Brusegan et al, ]; so that, Karavitakis et al [] have suggested its potential implication in the muscular hypotonia seen in patients with dose alterations at 3q13.2–q13.31 [Karavitakis et al, ], a phenotypic manifestation observed in our patient who also has only one copy of CCDC80 .…”
Section: Discussionmentioning
confidence: 69%
“…1). A previous description and the alignment of the predicted zebrafish proteins Ccdc80 (Chr9:35,060,460–35,084,513), Ccdc80‐like1 (ccdc80‐l1, Chr6:16,322,724–16,342,517) and Ccdc80‐like2 (ccdc80‐l2, Chr21: 18,662,129–18,669,986) has been performed by Brusegan and colleagues (Brusegan et al, ).…”
Section: Resultsmentioning
confidence: 99%
“…In the developing zebrafish expression of Dro1/Ccdc80 and Ccdc80-like1, a gene which displays 44 % sequence identity with human DRO1/CCDC80, has been shown to be positively regulated by the Hedgehog pathway [17,21]. Here, Dro1/Ccdc80 as well as Ccdc80-like1 became significantly downregulated after experimental or genetic inhibition of the Hedgehog signaling cascade, indicating a role as downstream targets of this pathway [17,21].…”
Section: Expression Pattern Of Dro1/ccdc80mentioning
confidence: 90%
“…Here, Dro1/Ccdc80 as well as Ccdc80-like1 became significantly downregulated after experimental or genetic inhibition of the Hedgehog signaling cascade, indicating a role as downstream targets of this pathway [17,21].…”
Section: Expression Pattern Of Dro1/ccdc80mentioning
confidence: 90%
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