Lily and Avraham Gildor Chair for the Investigation of Growth Factors, Elton Laboratory for Neuroendocrinology, Department of Human Molecular Genetics and Biochemistry, Sackler Faculty of Medicine, Sagol School of Neuroscience and Adams Super Center for Brain Studies, Tel Aviv University, Tel Aviv, Israel.
The Alfredo Federico Strauss Center for Computational Neuroimaging, Tel Aviv University, Tel Aviv, Israel.
Transl Psychiatry. 2019 Jan 15;9(1):2. doi: 10.1038/s41398-018-0357-6.
Activity-dependent neuroprotective protein (ADNP), essential for brain formation, was discovered as a leading de novo mutated gene causing the autism-like ADNP syndrome. This syndrome is phenotypically characterized by global developmental delays, intellectual disabilities, speech impediments, and motor dysfunctions. The Adnp haploinsufficient mouse mimics the human ADNP syndrome in terms of synapse density and gene expression patterns, as well as in developmental, motor, and cognitive abilities. Peripheral ADNP was also discovered as a biomarker for Alzheimer's disease and schizophrenia, with nasal administration of the ADNP snippet peptide NAP (enhancing endogenous ADNP activity) leading to partial cognitive and functional protection at the cellular, animal and clinical settings. Here, a novel formulation for effective delivery of NAP is provided with superior brain penetration capabilities. Also provided are methods for treating pertinent clinical implications such as autism, cognitive impairments, olfactory deficits, and muscle strength using the formulation in the Adnp haploinsufficient mouse. Results showed a dramatically specific increase in brain/body bioavailability with the new formulation, without breaching the blood brain barrier. Additional findings included improvements using daily intranasal treatments with NAP, at the behavioral and brain structural levels, diffusion tensor imaging (DTI), translatable to clinical practice. Significant effects on hippocampal and cerebral cortical expression of the presynaptic Slc17a7 gene encoding vesicular excitatory glutamate transporter 1 (VGLUT1) were observed at the RNA and immunohistochemical levels, explaining the DTI results. These findings tie for the first time a reduction in presynaptic glutamatergic synapses with the autism/Alzheimer's/schizophrenia-linked ADNP deficiency coupled with amelioration by NAP (CP201).
活性依赖型神经保护蛋白(ADNP)是大脑形成所必需的,它被发现是导致类似自闭症的 ADNP 综合征的主要从头突变基因。该综合征的表型特征为全面发育迟缓、智力障碍、言语障碍和运动功能障碍。Adnp 半不足小鼠在突触密度和基因表达模式以及发育、运动和认知能力方面模拟人类 ADNP 综合征。外周 ADNP 也被发现是阿尔茨海默病和精神分裂症的生物标志物,ADNP 短肽 NAP(增强内源性 ADNP 活性)的鼻内给药导致细胞、动物和临床水平的部分认知和功能保护。在这里,提供了一种用于有效传递 NAP 的新型制剂,具有卓越的脑穿透能力。还提供了使用 Adnp 半不足小鼠中的制剂治疗相关临床意义(如自闭症、认知障碍、嗅觉缺陷和肌肉力量)的方法。结果表明,与新配方相比,大脑/身体生物利用度显著增加,而不会突破血脑屏障。其他发现包括使用 NAP 进行每日鼻内治疗,在行为和大脑结构水平上,扩散张量成像(DTI),可转化为临床实践。在 RNA 和免疫组织化学水平上观察到编码囊泡兴奋性谷氨酸转运体 1(VGLUT1)的突触前 Slc17a7 基因的海马和大脑皮质表达显著增加,这解释了 DTI 结果。这些发现首次将与自闭症/阿尔茨海默病/精神分裂症相关的 ADNP 缺乏症与 NAP(CP201)改善相关的突触前谷氨酸能突触减少联系起来。