Characterization of a naturally occurring mutation V368M in the human glucagon receptor and its association with metabolic disorders

  • Biochem J. 2020 Jul 17;477(13):2581-2594. doi: 10.1042/BCJ20200235.
Guangyao Lin  #  1  2  3 Qiaofeng Liu  #  4 Antao Dai  1 Xiaoqing Cai  1 Qingtong Zhou  5 Xi Wang  1  3 Yan Chen  4 Chenyu Ye  4 Jie Li  1  3 Dehua Yang  1  3 Ming-Wei Wang  1  2  3  4  6
Affiliations
  • 1. The National Center for Drug Screening and CAS Key Laboratory of Receptor Research, Shanghai Institute of Materia Medica, Chinese Academy of Sciences (CAS), Shanghai 201203, China.
  • 2. School of Life Science and Technology, ShanghaiTech University, Shanghai 201210, China.
  • 3. University of Chinese Academy of Sciences, Beijing 100049, China.
  • 4. School of Pharmacy, Fudan University, Shanghai 201203, China.
  • 5. iHuman Institute, ShanghaiTech University, Shanghai 201210, China.
  • 6. School of Basic Medical Sciences, Fudan University, Shanghai 200032, China.
  • # Contributed equally.
Abstract

Glucagon is a peptide hormone secreted by islet α cells. It plays crucial roles in glucose homeostasis and metabolism by activating its cognate Glucagon Receptor (GCGR). A naturally occurring deleterious mutation V368M in the human GCGR leads to reduced ligand binding and down-regulation of glucagon signaling. To examine the association between this mutation and metabolic disorders, a knock-in mouse model bearing homozygous V369M substitution (equivalent to human V368M) in GCGR was made using CRISPR-Cas9 technology. These GcgrV369M+/+ mice displayed lower fasting blood glucose levels with improved glucose tolerance compared with wild-type controls. They also exhibited hyperglucagonemia, pancreas enlargement and α cell hyperplasia with a lean phenotype. Additionally, V369M mutation resulted in a reduction in adiposity with normal body weight and food intake. Our findings suggest a key role of V369M/V368M mutation in GCGR-mediated glucose homeostasis and pancreatic functions, thereby pointing to a possible interplay between GCGR defect and metabolic disorders.

Keywords
V368M; glucagon receptor; metabolic disorders; mouse model; mutation.