Proceedings of the National Academy of Sciences · 2008 · 561 citations · 42 references
Life SciencesGeneticsNeuroscienceAutism Spectrum DisordersMolecular NeuroscienceNeurogeneticsAutism Spectrum Disorder GeneticsNeurodevelopmental DisordersRestricted BehaviorsSynaptic DysfunctionSynaptic TransmissionSynaptogenesisAutism Spectrum Disorder ModelsGenetic Disease ModelsLoss-of-function MutationsNeuroligin GenesSingle-gene MutationsSynaptic Cell AdhesionSynaptic Gene MutationsUltrasonic Communication
Autism spectrum conditions are heritable disorders marked by impaired social interaction, language deficits, and repetitive behaviors, and even single‑gene mutations can cause monogenic forms such as loss‑of‑function mutations in synaptic regulators like NLGN4. Nlgn4 knockout mice exhibit selective deficits in reciprocal social interactions and communication that mirror human ASCs, demonstrating that synaptic maturation networks are central to ASC susceptibility and validating these mice as models for studying ASC neurobiology.
Autism spectrum conditions (ASCs) are heritable conditions characterized by impaired reciprocal social interactions, deficits in language acquisition, and repetitive and restricted behaviors and interests. In addition to more complex genetic susceptibilities, even mutation of a single gene can lead to ASC. Several such monogenic heritable ASC forms are caused by loss-of-function mutations in genes encoding regulators of synapse function in neurons, including NLGN4. We report that mice with a loss-of-function mutation in the murine NLGN4 ortholog Nlgn4, which encodes the synaptic cell adhesion protein Neuroligin-4, exhibit highly selective deficits in reciprocal social interactions and communication that are reminiscent of ASCs in humans. Our findings indicate that a protein network that regulates the maturation and function of synapses in the brain is at the core of a major ASC susceptibility pathway, and establish Neuroligin-4-deficient mice as genetic models for the exploration of the complex neurobiological disorders in ASCs.
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