Searching for biomarkers of CDKL5 disorder: early-onset visual impairment in CDKL5 mutant mice.

ABSTRACT: CDKL5 disorder is a neurodevelopmental disorder still without a cure. Murine models of CDKL5 disorder have been recently generated raising the possibility of preclinical testing of treatments. However, unbiased, quantitative biomarkers of high translational value to monitor brain function are still missing. Moreover, the analysis of treatment is hindered by the challenge of repeatedly and non-invasively testing neuronal function. We analyzed the development of visual responses in a mouse model of CDKL5 disorder to introduce visually evoked responses as a quantitative method to assess cortical circuit function. Cortical visual responses were assessed in CDKL5 null male mice, heterozygous females, and their respective control wild-type littermates by repeated transcranial optical imaging fro

SUBMITTER: Mazziotti R 

PROVIDER: S-EPMC5458338 | biostudies-literature | 2017 Jun

REPOSITORIES: biostudies-literature