Nature · 2011
Mutations causing syndromic autism define an axis of synaptic pathophysiology.
СтатьяПоддержка NIH: внешняя программаПоддержка исследования: вне правительства СШАПоддержка правительства СШАНазвание и аннотация: Перевод в очереди
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Auerbach Benjamin D, Osterweil Emily K, Bear Mark F
Аннотация
Tuberous sclerosis complex and fragile X syndrome are genetic diseases characterized by intellectual disability and autism. Because both syndromes are caused by mutations in genes that regulate protein synthesis in neurons, it has been hypothesized that excessive protein synthesis is one core pathophysiological mechanism of intellectual disability and autism. Using electrophysiological and biochemical assays of neuronal protein synthesis in the hippocampus of Tsc2(+/-) and Fmr1(-/y) mice, here we show that synaptic dysfunction caused by these mutations actually falls at opposite ends of a physiological spectrum. Synaptic, biochemical and cognitive defects in these mutants are corrected by treatments that modulate metabotropic glutamate receptor 5 in opposite directions, and deficits in the mutants disappear when the mice are bred to carry both mutations. Thus, normal synaptic plasticity and cognition occur within an optimal range of metabotropic glutamate-receptor-mediated protein synthesis, and deviations in either direction can lead to shared behavioural impairments.
Предметные рубрики MeSH
AnimalsAutistic DisorderDisease Models, AnimalElectrical SynapsesFemaleFragile X Messenger Ribonucleoprotein 1Gene Expression RegulationHippocampusMaleMiceMice, Inbred C57BLMutationReceptor, Metabotropic Glutamate 5Receptors, Metabotropic GlutamateSyndromeTOR Serine-Threonine KinasesTuberous Sclerosis Complex 2 ProteinTumor Suppressor Proteins