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© British Veterinary Association 2020. No commercial re-use. See rights and permissions. Published by BMJ.Oligophrenin-1 (Ophn1) encodes a Rho GTPase activating protein whose mutations cause X-linked intellectual disability (XLID) in humans. Loss of function of Ophn1 leads to impairments in the maturation and function of excitatory and inhibitory synapses, causing deficits in synaptic structure, function and plasticity. Epilepsy is a frequent co-morbidity in patients with Ophn1-dependent XLID, but the cellular bases of hyperexcitability