Assessment of Mendelian and risk-factor genes in Alzheimer disease: A prospective nationwide clinical utility study and recommendations for genetic screening

Genet Med. 2024 May;26(5):101082. doi: 10.1016/j.gim.2024.101082. Epub 2024 Jan 24.

Abstract

Purpose: To assess the likely pathogenic/pathogenic (LP/P) variants rates in Mendelian dementia genes and the moderate-to-strong risk factors rates in patients with Alzheimer disease (AD).

Methods: We included 700 patients in a prospective study and performed exome sequencing. A panel of 28 Mendelian and 6 risk-factor genes was interpreted and returned to patients. We built a framework for risk variant interpretation and risk gradation and assessed the detection rates among early-onset AD (EOAD, age of onset (AOO) ≤65 years, n = 608) depending on AOO and pedigree structure and late-onset AD (66 < AOO < 75, n = 92).

Results: Twenty-one patients carried a LP/P variant in a Mendelian gene (all with EOAD, 3.4%), 20 of 21 affected APP, PSEN1, or PSEN2. LP/P variant detection rates in EOAD ranged from 1.7% to 11.6% based on AOO and pedigree structure. Risk factors were found in 69.5% of the remaining 679 patients, including 83 (12.2%) being heterozygotes for rare risk variants, in decreasing order of frequency, in TREM2, ABCA7, ATP8B4, SORL1, and ABCA1, including 5 heterozygotes for multiple rare risk variants, suggesting non-monogenic inheritance, even in some autosomal-dominant-like pedigrees.

Conclusion: We suggest that genetic screening should be proposed to all EOAD patients and should no longer be prioritized based on pedigree structure.

Keywords: Alzheimer disease; Clinical utility; Exome; Pathogenic variant; Risk variant.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, N.I.H., Extramural

MeSH terms

  • Age of Onset
  • Aged
  • Aged, 80 and over
  • Alzheimer Disease* / diagnosis
  • Alzheimer Disease* / genetics
  • Amyloid beta-Protein Precursor / genetics
  • Exome Sequencing*
  • Female
  • Genetic Predisposition to Disease*
  • Genetic Testing* / methods
  • Humans
  • Male
  • Membrane Glycoproteins*
  • Middle Aged
  • Pedigree
  • Presenilin-1 / genetics
  • Presenilin-2* / genetics
  • Prospective Studies
  • Receptors, Immunologic*
  • Risk Factors

Substances

  • Presenilin-2
  • PSEN2 protein, human
  • Presenilin-1
  • PSEN1 protein, human
  • Amyloid beta-Protein Precursor
  • APP protein, human
  • TREM2 protein, human
  • Membrane Glycoproteins
  • Receptors, Immunologic