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A mother and her daughter carrying a pathogenic expansion of the HTT gene with a phenotype encompassing motor neuron disease and Huntington's disease.

Antonio CanosaSara CabrasFrancesca Di PedeUmberto ManeraRosario VastaCristina MogliaAndrea CalvoSalvatore GalloneAdriano Chiò
Published in: Clinical genetics (2023)
Recently, pathogenic expansions (range 40-64 CAG repeats) in the HTT gene have been found in patients diagnosed with pure frontotemporal dementia/amyotrophic lateral sclerosis (FTD/ALS). We report a mother with Huntington's disease (HD) associated with motor neuron disease (MND) signs and her daughter suffering from ALS with subtle signs of HD, both carrying a pathogenic allele of the HTT gene (i.e., >39 repeats). The co-occurrence of MND and chorea has been reported in previous cases. Subjects showing both ALS and HD signs and carrying HTT pathogenic expansions in two generations of the same kindred have never been reported so far. The study of the overlap of disease mechanisms at the cellular level between TDP-43 and Huntingtin is relevant in an era offering promising strategies of targeted treatments in neurodegenerative disorders.
Keyphrases
  • amyotrophic lateral sclerosis
  • end stage renal disease
  • genome wide
  • copy number
  • healthcare
  • chronic kidney disease
  • gene expression
  • drug delivery
  • prognostic factors
  • cancer therapy
  • patient reported