Detection of the SQSTM1 Mutation in a Patient with Early-Onset Hippocampal Amnestic Syndrome

J Alzheimers Dis. 2021;79(2):477-481. doi: 10.3233/JAD-201231.

Abstract

Genetics has a major role in early-onset dementia, but the correspondence between genotype and phenotype is largely tentative. We describe a 54-year-old with familial early-onset slowly-progressive episodic memory impairment with the P392L-variant in SQSTM1. The patient showed cortical atrophy and hypometabolism in the temporal lobes, but no amyloidosis biomarkers. As symptoms/neuroimaging were suggestive for Alzheimer's disease-but biomarkers were not-and considering the family-history, genetic analysis was performed, revealing the P392L-variant in SQSTM1, which encodes for sequestosome-1/p62. Increasing evidence suggests a p62 involvement in neurodegeneration and SQSTM1 mutations have been found to cause amyotrophic lateral sclerosis/frontotemporal dementia. Our report suggests that the clinical spectrum of SQSTM1 variants is wider.

Keywords: Alzheimer’s disease; SQSTM1; early-onset dementia; next-generation sequencing; p62.

Publication types

  • Case Reports

MeSH terms

  • Age of Onset
  • Amnesia / genetics*
  • Amyotrophic Lateral Sclerosis / genetics*
  • Frontotemporal Dementia / genetics*
  • Genetic Testing
  • Genotype
  • High-Throughput Nucleotide Sequencing
  • Hippocampus*
  • Humans
  • Male
  • Middle Aged
  • Mutation*
  • Sequestosome-1 Protein / genetics*

Substances

  • SQSTM1 protein, human
  • Sequestosome-1 Protein

Supplementary concepts

  • Frontotemporal Dementia With Motor Neuron Disease