disease 1,732 words KG: ent-dise-80d07a60
Contents

spinocerebellar-ataxia-type-1

Disease Info
CAG repeat expansionThe pathogenic mutation is an expanded CAG trinucleotide repeat in the ATXN1 gene on chromosome 6p22-23
Normal alleles6-35 CAG repeats
Intermediate alleles (premutation)36-38 repeats (may expand in meiosis)
Full mutation alleles39-83+ repeats (disease-causing)
AnticipationLarger repeats correlate with earlier onset and more severe disease
Normal functionAtaxin-1 is a transcriptional regulator involved in RNA splicing and neuronal gene expression
Pathogenic mechanismThe expanded polyglutamine tract causes toxic gain-of-function, leading to protein misfolding, aggregation, and cellular dysfunction
Selective vulnerabilityPurkinje cells in the cerebellum and neurons in the inferior olive are particularly susceptible to ataxin-1 toxicity
Cerebellar atrophySevere loss of Purkinje cells and granular layer neurons
Inferior olivary nucleus degenerationNeuronal loss in the inferior olive
Brainstem involvementDegeneration of cranial nerve nuclei
Spinal cord pathologyLoss of neurons in the posterior columns and anterior horns
DatabasesOMIMOrphanetClinicalTrialsPubMed

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