FMR1 Carriers Report Executive Function Changes Prior to Fragile X-Associated Tremor/Ataxia Syndrome: A Longitudinal Study.

ABSTRACT:

Background

Men with fragile X-associated tremor/ataxia syndrome (FXTAS) often develop executive dysfunction, characterized by disinhibition, frontal dyscontrol of movement, and working memory and attention changes. Although cross-sectional studies have suggested that earlier executive function changes may precede FXTAS, the lack of longitudinal studies has made it difficult to address this hypothesis.

Objective

To determine whether executive function deterioration experienced by premutation carriers (PC) in daily life precedes and predicts FXTAS.

Methods

This study included 66 FMR1 PC ranging from 40 to 78 years (mean, 59.5) and 31 well-matched healthy controls (HC) ages 40 to 75 (mean, 57.7) at baseline. Eighty-four participants returned for 2 to 5 follow up visits o

SUBMITTER: Hessl D 

PROVIDER: S-EPMC11268876 | biostudies-literature | 2024 Mar

REPOSITORIES: biostudies-literature