Mohammad Shahid, Wang Li, Torii Masaaki, Hashimoto-Torii Kazue
Center for Neuroscience Research, Children's Research Institute, Children's National Hospital, Washington, DC, United States.
Department of Pediatrics, Pharmacology and Physiology, School of Medicine and Health Sciences, George Washington University, Washington, DC, United States.
Int J Neuropsychopharmacol. 2025 Sep 1;28(9). doi: 10.1093/ijnp/pyaf055.
Fetal alcohol spectrum disorders (FASD) show a myriad of cognitive and neurological deficits, with the prevalence estimated to be 1% to 5 % in children. To date, there are no effective treatments for these deficits in FASD. In a mouse model of FASD, daily intraperitoneal administration of a potassium calcium-activated channel subfamily N member 2 (KCNN2) blocking peptide has been shown to improve motor learning deficits due to upregulation of KCNN2 channels. This study investigates whether intranasal administration of a KCNN2 blocking peptide, Leiurotoxin-1 Dab7 (Lei-Dab7), can improve cognitive flexibility, specifically reversal learning deficits, in these mice.
We utilized a mouse model of prenatal alcohol exposure. Cognitive flexibility was assessed using the water T-maze test at postnatal day 40. Lei-Dab7's specificity and cytotoxicity were evaluated in vitro, and intranasal delivery efficiency was confirmed through immunohistochemistry, quantifying its distribution and binding to neurons with elevated KCNN2 expression in the prefrontal cortex.
Lei-Dab7 showed high specificity and negligible cytotoxicity in vitro. Intranasal administration efficiently delivered Lei-Dab7 to the prefrontal cortex, where it specifically bound to neurons expressing increased KCNN2 channels. Behavioral tests demonstrated that Lei-Dab7 significantly improved cognitive flexibility, reversing the deficits in the water T-maze test seen in ethanol-exposed mice, without apparent acute physiological adverse effects.
Intranasal administration of KCNN2 blockers, such as Lei-Dab7, represents a promising, non-invasive therapeutic approach for treating cognitive inflexibility and possibly other cognitive dysfunctions associated with FASD.
胎儿酒精谱系障碍(FASD)表现出众多认知和神经功能缺陷,据估计儿童中的患病率为1%至5%。迄今为止,针对FASD中的这些缺陷尚无有效的治疗方法。在FASD的小鼠模型中,每日腹腔注射钾钙激活通道亚家族N成员2(KCNN2)阻断肽已显示可改善因KCNN2通道上调而导致的运动学习缺陷。本研究调查鼻内给予KCNN2阻断肽Leiurotoxin - 1 Dab7(Lei - Dab7)是否能改善这些小鼠的认知灵活性,特别是逆向学习缺陷。
我们使用了产前酒精暴露的小鼠模型。在出生后第40天使用水T迷宫试验评估认知灵活性。在体外评估Lei - Dab7的特异性和细胞毒性,并通过免疫组织化学确认鼻内递送效率,量化其在额叶前皮质中与KCNN2表达升高的神经元的分布和结合情况。
Lei - Dab7在体外显示出高特异性和可忽略不计的细胞毒性。鼻内给药有效地将Lei - Dab7递送至额叶前皮质,在那里它特异性地与表达增加的KCNN2通道的神经元结合。行为测试表明,Lei - Dab7显著改善了认知灵活性,逆转了乙醇暴露小鼠在水T迷宫试验中出现的缺陷,且无明显的急性生理不良反应。
鼻内给予KCNN2阻断剂,如Lei - Dab7,代表了一种有前景的、非侵入性的治疗方法,用于治疗认知灵活性障碍以及可能与FASD相关的其他认知功能障碍。