Phenotypic variation of FXN compound heterozygotes in a Friedreich ataxia cohort.
Megan M ShenChristian RummeyDavid R LynchPublished in: Annals of clinical and translational neurology (2024)
These data support that the typical FRDA phenotype is driven by frataxin deficiency, especially severe in compound heterozygotes with minimal/no function mutations, whereas the heterogeneous presentations of those with partial function mutations may indicate other contributing factors to FRDA pathogenesis.