FXS_FMR1

Gene
FMR1
Disease
FXS
Inheritance
XD
Classification
Definitive
Total Score
14.5
Publications Reviewed
8
Publication Span
28.67 years
Last Updated
8/18/2025
Curator(s)
Macayla Weiner

Note: Maximum score caps apply at evidence type, category, and supercategory levels, so section totals may be lower than the raw sum of row scores.

Genetic evidence

Total: 11

Singular EvidenceProbandsPMID:3246354262362 probands from 2 cohorts
StatisticsCase-Control DataPMID:264914885statistically significant, no bias, not well matched
2 rows

Experimental evidence

Total: 3.5

FunctionBiochemical functionPMID:313424420.5transcriptional silencing of gene
FunctionProtein interactionPMID:13424420.5absence of [FMRP] evokes FXS; "associated marked decrease in... protein product"
FunctionRegulatory ImpactPMID:306420660.5It is believed that only a small number of embryonic progenitor cells will go on to form the brain, so the ratio of cells that have the affected X active to silenced is thought to significantly affect the level of FMRP expression in the developing central nervous system.
Functional AlterationPatient cellsPMID:306780240.5leads to abnormal methylation of MFR1
ModelsNon-human model organismPMID:298681081257 patients
ModelsCell culturePMID:252874580.5control humans
6 rows