English (United Kingdom)

https://curated-unify.zendy.io/wp-json/zendy-region/v1/featured_content/oa?rat=en

https://curated-unify.zendy.io/wp-json/zendy-region/v1/highlighted_journal/

Global: Zendy Tools annual

Presents the keyphrase highlighting as premium feature

Keyphrase Highlighting

Presents the summarisation as premium feature

Summarisation

Insights

Presents the pdf analysis as premium feature

PDF Analysis

Presents the zaia usage as premium feature

ZAIA

Global: Zendy Tools monthly

Global: Zendy Plus monthly

Presents the access of premium content as premium feature

Premium Content

Global: Zendy Plus annual

Global: Zendy Free Plan

foxP2 , a forkhead-domain transcription factor, is critical for speech and language development in humans, but its role in the establishment of CNS connectivity is unclear. While  in vitro  studies have identified axon guidance molecules as targets of  foxP2  regulation, and cell culture assays suggest a role for  foxP2  in neurite outgrowth,  in vivo  studies have been lacking regarding a role for  foxP2  in axon pathfinding. We used a modified zinc finger nuclease methodology to generate mutations in the zebrafish  foxP2  gene. Using PCR-based high resolution melt curve analysis (HRMA) of G0 founder animals, we screened and identified three mutants carrying nonsense mutations in the 2 nd  coding exon: a 17 base-pair (bp) deletion, an 8bp deletion, and a 4bp insertion. Sequence analysis of cDNA confirmed that these were frameshift mutations with predicted early protein truncations. Homozygous mutant fish were viable and fertile, with unchanged body morphology, and no apparent differences in CNS apoptosis, proliferation, or patterning at embryonic stages. There was a reduction in expression of the known  foxP2  target gene  cntnap2  that was rescued by injection of wild-type  foxP2  transcript. When we examined axon pathfinding using a pan-axonal marker or transgenic lines, including a  foxP2 -neuron-specific enhancer, we did not observe any axon guidance errors. Our findings suggest that  foxP2  is not necessary for axon pathfinding during development.

Zebrafish foxP2 Zinc Finger Nuclease Mutant Has Normal Axon Pathfinding