Abstract
Fibroblast growth factor 14 (FGF14) belongs to a distinct subclass of FGFs that is expressed in the developing and adult CNS. We disrupted the Fgf14 gene and introduced an Fgf14N-β-Gal allele that abolished Fgf14 expression and generated a fusion protein (FGF14N-β-gal) containing the first exon of FGF14 and β-galactosidase. Fgf14-deficient mice were viable, fertile, and anatomically normal, but developed ataxia and a paroxysmal hyperkinetic movement disorder. Neuropharmacological studies showed that Fgf14-deficient mice have reduced responses to dopamine agonists. The paroxysmal hyperkinetic movement disorder phenocopies a form of dystonia, a disease often associated with dysfunction of the putamen. Strikingly, the FGF14N-β-gal chimeric protein was efficiently transported into neuronal processes in the basal ganglia and cerebellum. Together, these studies identify a novel function for FGF14 in neuronal signaling and implicate FGF14 in axonal trafficking and synaptosomal function.
Original language | English |
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Pages (from-to) | 25-38 |
Number of pages | 14 |
Journal | Neuron |
Volume | 35 |
Issue number | 1 |
DOIs | |
State | Published - Jul 3 2002 |