Zebrafish Chromosome 14 Gene Differential Expression in the fmr1 ^{h u2787} Model of Fragile X Syndrome.

ABSTRACT: Zebrafish represent a valuable model for investigating the molecular and cellular basis of Fragile X syndrome (FXS). Reduced expression of the zebrafish FMR1 orthologous gene, fmr1, causes developmental and behavioural phenotypes related to FXS. Zebrafish homozygous for the hu2787 non-sense mutation allele of fmr1 are widely used to model FXS, although FXS-relevant phenotypes seen from morpholino antisense oligonucleotide (morpholino) suppression of fmr1 transcript translation were not observed when hu2787 was first described. The subsequent discovery of transcriptional adaptation (a form of genetic compensation), whereby mutations causing non-sense-mediated decay of transcripts can drive compensatory upregulation of homologous transcripts independent of protein

SUBMITTER: Barthelson K 

PROVIDER: S-EPMC8203322 | biostudies-literature | 2021

REPOSITORIES: biostudies-literature