A Particular SORL1 Micro-haplotype May Prevent Severe Liver Disease in a French Cohort of Alpha 1-Antitrypsin-deficient Children

J Pediatr Gastroenterol Nutr. 2021 Sep 1;73(3):e68-e72. doi: 10.1097/MPG.0000000000003125.

Abstract

The presence of modifier genes is now well recognized in severe liver disease outcome associated with alpha-1-antitrypsin deficiency (A1ATD) but their identification remains to be fully elucidated. To address this goal, we performed a candidate gene study with the SORL1 gene, already identified as risk gene in early-onset Alzheimer Disease families. A particular SORL1 micro-haplotype constituted with 3 SNPs (wild-type form TTG) was genotyped on 86 ZZ A1ATD children issued from 66 families. Interestingly, the mutated forms of this micro-haplotype (CAT most of the time) were associated with lower occurrence of severe liver disease and in cellulo studies showed that SORL1 influences Z-A1ATD cellular toxicity and biogenesis. These data suggest that the mutated CAT form of SORL1 micro-haplotype may partly prevent from severe liver disease in A1ATD children. Overall, these findings support a replication study on an independent cohort and additional in cellulo studies to confirm these promising results.

Trial registration: ClinicalTrials.gov NCT01862211.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Child
  • Cohort Studies
  • France
  • Haplotypes
  • Humans
  • LDL-Receptor Related Proteins* / genetics
  • Liver Diseases / genetics*
  • Membrane Transport Proteins* / genetics
  • Polymorphism, Single Nucleotide
  • alpha 1-Antitrypsin Deficiency* / genetics
  • alpha 1-Antitrypsin* / genetics

Substances

  • LDL-Receptor Related Proteins
  • Membrane Transport Proteins
  • SORL1 protein, human
  • alpha 1-Antitrypsin

Associated data

  • ClinicalTrials.gov/NCT01862211