Novel and ultra-rare damaging variants in neuropeptide signaling are associated with disordered eating behaviors

PLoS One. 2017 Aug 28;12(8):e0181556. doi: 10.1371/journal.pone.0181556. eCollection 2017.

Abstract

Objective: Eating disorders develop through a combination of genetic vulnerability and environmental stress, however the genetic basis of this risk is unknown.

Methods: To understand the genetic basis of this risk, we performed whole exome sequencing on 93 unrelated individuals with eating disorders (38 restricted-eating and 55 binge-eating) to identify novel damaging variants. Candidate genes with an excessive burden of predicted damaging variants were then prioritized based upon an unbiased, data-driven bioinformatic analysis. One top candidate pathway was empirically tested for therapeutic potential in a mouse model of binge-like eating.

Results: An excessive burden of novel damaging variants was identified in 186 genes in the restricted-eating group and 245 genes in the binge-eating group. This list is significantly enriched (OR = 4.6, p<0.0001) for genes involved in neuropeptide/neurotrophic pathways implicated in appetite regulation, including neurotensin-, glucagon-like peptide 1- and BDNF-signaling. Administration of the glucagon-like peptide 1 receptor agonist exendin-4 significantly reduced food intake in a mouse model of 'binge-like' eating.

Conclusions: These findings implicate ultra-rare and novel damaging variants in neuropeptide/neurotropic factor signaling pathways in the development of eating disorder behaviors and identify glucagon-like peptide 1-receptor agonists as a potential treatment for binge eating.

MeSH terms

  • Adolescent
  • Adult
  • Appetite Regulation / genetics*
  • Computational Biology
  • Exome
  • Feeding and Eating Disorders / genetics*
  • Feeding and Eating Disorders / metabolism
  • Female
  • Gene Regulatory Networks*
  • Genetic Predisposition to Disease
  • Humans
  • Male
  • Middle Aged
  • Neuropeptides / genetics*
  • Neuropeptides / metabolism
  • Signal Transduction / physiology*
  • Young Adult

Substances

  • Neuropeptides