Cited by 86Genetics and Neurodevelopmental DisordersAutism Spectrum Disorder ResearchNeuroscience and Neuropharmacology Research

Neuroligin-deficient mutants of C. elegans have sensory processing deficits and are hypersensitive to oxidative stress and mercury toxicity

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Hunter, Jerrod W., Mullen, Gregory P., McManus, John R., Heatherly, Jessica M., Duke, Angie, Rand, James B.·2010·Disease Models & Mechanisms

Abstract

SUMMARY: Neuroligins are postsynaptic cell adhesion proteins that bind specifically to presynaptic membrane proteins called neurexins. Mutations in human neuroligin genes are associated with autism spectrum disorders in some families. The nematode Caenorhabditis elegans has a single neuroligin gene (nlg-1), and approximately a sixth of C. elegans neurons, including some sensory neurons, interneurons and a subset of cholinergic motor neurons, express a neuroligin transcriptional reporter. Neuroligin-deficient mutants of C. elegans are viable, and they do not appear deficient in any major motor functions. However, neuroligin mutants are defective in a subset of sensory behaviors and sensory processing, and are hypersensitive to oxidative stress and mercury compounds; the behavioral deficits are strikingly similar to traits frequently associated with autism spectrum disorders. Our results suggest a possible link between genetic defects in synapse formation or function, and sensitivity to environmental factors in the development of autism spectrum disorders.

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Crossref · 10.1242/dmm.003442

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Authors
Hunter, J. W., Mullen, G. P., McManus, J. R., Heatherly, J. M., Duke, A., & Rand, J. B.
Affiliations
Oklahoma Medical Research Foundation (US)
Corresponding author
Rand, James B.
Venue
Disease Models & Mechanisms
Published
2010-04-28
Volume
3
Issue
5-6
Pages
366-376
ISSN
  • 1754-8403 (ISSN-L)
  • 1754-8411
OpenAlex
W2146735224
Cited by
86
References
94
Citations by year
2026: 2 · 2025: 4 · 2024: 3 · 2023: 1 · 2022: 5 · 2021: 5 · 2020: 10 · 2019: 3 · 2018: 4 · 2017: 5 · 2016: 1 · 2015: 9 · 2014: 8 · 2013: 11 · 2012: 5
FWCI
4.67
Type
journal article
Language
en
Open access
Locations
  • Disease Models & Mechanisms (publishedVersion)
  • PubMed Central (publishedVersion)
  • PubMed (publishedVersion)
  • HighWire Press Open Archive (submittedVersion)
Funders
  • Autism Speaks (AS1975)
  • Ludwig Institute for Cancer Research
Topics
  • Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
  • Life Sciences › Neuroscience › Cognitive Neuroscience › Autism Spectrum Disorder Research
  • Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Neuroscience and Neuropharmacology Research
Keywords
neuroligins, Caenorhabditis elegans, sensory processing deficits, sensory behavior, oxidative stress sensitivity, mercury toxicity, autism spectrum disorder, neurexin, synaptogenesis, environmental sensitivity
MeSH terms
Animals, Behavior, Animal, Cues, Humans, Mercury, Mutation, Neurons, Recombinant Fusion Proteins, Sensation, Synapses, Temperature, Biomarkers, Cell Adhesion Molecules, Neuronal, Caenorhabditis elegans, Sequence Homology, Amino Acid, Genes, Reporter, Oxidative Stress, Protein Transport, Muscle Cells

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