Targeting the site encoded by SERPINA1*E342K for treating alpha-1 antitrypsin deficiency-associated liver diseases

FEBS Lett. 2019 Jul;593(14):1849-1862. doi: 10.1002/1873-3468.13452. Epub 2019 Jun 6.

Abstract

Alpha1-antitrypsin (AAT) deficiency predisposes individuals to emphysema and liver diseases such as cirrhosis and hepatocellular carcinoma. The deficiency results from mutations in the SERPIN1A gene encoding AAT molecules that cause hepatotoxic retention within the endoplasmic reticulum. Since the E342K mutation is the basis for destabilization leading to lung and liver pathologies, we used the crystal structure of the mutated AAT as the basis for molecular docking selection of candidate compounds that may bind and stabilize the 342K structural pocket. We identified compounds that inhibited intracellular accumulation of AAT in hepatocytes in vitro. These data suggest that drug binding to a structural site encoded by a mutation associated with AAT deficiency has the potential for clinical utility by modulating conformational transitions.

Keywords: alpha-1 antitrypsin; liver disease; molecular docking; structure-based drug discovery.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cell Line
  • Drug Evaluation, Preclinical
  • Humans
  • Intracellular Space / drug effects
  • Intracellular Space / metabolism
  • Liver Diseases / complications*
  • Liver Diseases / drug therapy*
  • Liver Diseases / genetics
  • Molecular Docking Simulation
  • Molecular Targeted Therapy*
  • Mutation*
  • Protein Conformation
  • alpha 1-Antitrypsin / chemistry
  • alpha 1-Antitrypsin / genetics*
  • alpha 1-Antitrypsin / metabolism
  • alpha 1-Antitrypsin Deficiency / complications*

Substances

  • Serpina1a protein, mouse
  • alpha 1-Antitrypsin