Investigating the Endo-Lysosomal System in Major Neurocognitive Disorders Due to Alzheimer's Disease, Frontotemporal Lobar Degeneration and Lewy Body Disease: Evidence for SORL1 as a Cross-Disease Gene

Int J Mol Sci. 2021 Dec 20;22(24):13633. doi: 10.3390/ijms222413633.

Abstract

Dysfunctions in the endo-lysosomal system have been hypothesized to underlie neurodegeneration in major neurocognitive disorders due to Alzheimer's disease (AD), Frontotemporal Lobar Degeneration (FTLD), and Lewy body disease (DLB). The aim of this study is to investigate whether these diseases share genetic variability in the endo-lysosomal pathway. In AD, DLB, and FTLD patients and in controls (948 subjects), we performed a targeted sequencing of the top 50 genes belonging to the endo-lysosomal pathway. Genetic analyses revealed (i) four previously reported disease-associated variants in the SORL1 (p.N1246K, p.N371T, p.D2065V) and DNAJC6 genes (p.M133L) in AD, FTLD, and DLB, extending the previous knowledge attesting SORL1 and DNAJC6 as AD- and PD-related genes, respectively; (ii) three predicted null variants in AD patients in the SORL1 (p.R985X in early onset familial AD, p.R1207X) and PPT1 (p.R48X in early onset familial AD) genes, where loss of function is a known disease mechanism. A single variant and gene burden analysis revealed some nominally significant results of potential interest for SORL1 and DNAJC6 genes. Our data highlight that genes controlling key endo-lysosomal processes (i.e., protein sorting/transport, clathrin-coated vesicle uncoating, lysosomal enzymatic activity regulation) might be involved in AD, FTLD and DLB pathogenesis, thus suggesting an etiological link behind these diseases.

Keywords: DNAJC6; NGS; PPT1; SORL1; allele dose effect; cross-disease; endo-lysosomal genes; loss of function; multicarrier.

MeSH terms

  • Aged
  • Aged, 80 and over
  • Alzheimer Disease / genetics
  • Alzheimer Disease / metabolism*
  • Female
  • Frontotemporal Lobar Degeneration / genetics
  • Frontotemporal Lobar Degeneration / metabolism*
  • Genetic Predisposition to Disease*
  • HSP40 Heat-Shock Proteins / genetics*
  • High-Throughput Nucleotide Sequencing
  • Humans
  • LDL-Receptor Related Proteins / genetics*
  • Lewy Body Disease / genetics
  • Lewy Body Disease / metabolism*
  • Lysosomes / metabolism
  • Male
  • Membrane Transport Proteins / genetics*
  • Middle Aged
  • Polymorphism, Single Nucleotide*
  • Sequence Analysis, DNA

Substances

  • DNAJC6 protein, human
  • HSP40 Heat-Shock Proteins
  • LDL-Receptor Related Proteins
  • Membrane Transport Proteins
  • SORL1 protein, human