Abstract
Neuroligin 3 (NLGN3) and neurexins (NRXNs) constitute a canonical transsynaptic cell-adhesion pair, which has been implicated in autism. In autism spectrum disorder (ASD) development of sociality can be impaired. However, the molecular mechanism underlying NLGN3-mediated social development is unclear. Here, we identify non-canonical interactions between NLGN3 and protein tyrosine phosphatase δ (PTPδ) splice variants, competing with NRXN binding. NLGN3-PTPδ complex structure revealed a splicing-dependent interaction mode and competition mechanism between PTPδ and NRXNs. Mice carrying a NLGN3 mutation that selectively impairs NLGN3-NRXN interaction show increased sociability, whereas mice where the NLGN3-PTPδ interaction is impaired exhibit impaired social behavior and enhanced motor learning, with imbalance in excitatory/inhibitory synaptic protein expressions, as reported in the Nlgn3 R451C autism model. At neuronal level, the autism-related Nlgn3 R451C mutation causes selective impairment in the non-canonical pathway. Our findings suggest that canonical and non-canonical NLGN3 pathways compete and regulate the development of sociality.
Publication types
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Research Support, Non-U.S. Gov't
MeSH terms
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Amino Acid Sequence
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Animals
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Autism Spectrum Disorder / genetics*
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Autism Spectrum Disorder / metabolism
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Behavior Rating Scale
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Calcium-Binding Proteins / chemistry
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Calcium-Binding Proteins / genetics
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Calcium-Binding Proteins / metabolism*
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Cell Adhesion Molecules, Neuronal / chemistry
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Cell Adhesion Molecules, Neuronal / genetics
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Cell Adhesion Molecules, Neuronal / metabolism*
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Disease Models, Animal
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Female
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HEK293 Cells
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Humans
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Male
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Membrane Proteins / chemistry
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Membrane Proteins / genetics
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Membrane Proteins / metabolism*
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Mice
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Mice, Knockout
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Mice, Transgenic
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Mutation
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Nerve Tissue Proteins / chemistry
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Nerve Tissue Proteins / genetics
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Nerve Tissue Proteins / metabolism*
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Neural Cell Adhesion Molecules / chemistry
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Neural Cell Adhesion Molecules / genetics
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Neural Cell Adhesion Molecules / metabolism*
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Neurons / metabolism*
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Protein Domains
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Protein Splicing
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Receptor-Like Protein Tyrosine Phosphatases, Class 2 / chemistry
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Receptor-Like Protein Tyrosine Phosphatases, Class 2 / genetics
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Receptor-Like Protein Tyrosine Phosphatases, Class 2 / metabolism*
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Recombinant Proteins
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Signal Transduction / genetics
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Signal Transduction / physiology
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Social Behavior
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Synapses / genetics
Substances
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Calcium-Binding Proteins
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Cell Adhesion Molecules, Neuronal
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Membrane Proteins
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Nerve Tissue Proteins
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Neural Cell Adhesion Molecules
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Nrxn1 protein, mouse
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Recombinant Proteins
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neuroligin 3
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Receptor-Like Protein Tyrosine Phosphatases, Class 2