The Cain Foundation Laboratories, The Jan and Dan Duncan Neurological Research Institute, Texas Children's Hospital, Houston, TX 77030.
Department of Pediatrics, Baylor College of Medicine, Houston, TX 77030.
eNeuro. 2017 Apr 28;4(2). doi: 10.1523/ENEURO.0014-17.2017. eCollection 2017 Mar-Apr.
Neurobehavioral abnormalities are commonly associated with intractable childhood epilepsy. Studies from numerous labs have demonstrated cognitive and socialization deficits in rats and mice that have experienced early-life seizures. However, the cellular and molecular mechanisms underlying these effects are unknown. Previously, experiments have shown that recurrent seizures in infancy suppress the growth of hippocampal dendrites at the same time they impair learning and memory. Experiments in slice cultures have also demonstrated dendrite growth suppression. Here, we crossed calcineurin B1 (CaNB1) floxed and Thy1GFP-M mice to produce mice that were homozygous for the both the floxed CaNB1 and the Thy1GFP-M transgene. Littermates that were homozygous for wild-type CaNB1 and Thy1GFP-M served as controls. Hippocampal slice cultures from these mice were transfected with an AAV/hSyn-mCherry-Cre virus to eliminate CaNB1 from neurons. Immunohistochemical results showed that CaNB1 was eliminated from at least 90% of the transfected CA1 pyramidal cells. Moreover, the CaN-dependent nuclear translocation of the CREB transcription coactivator, CREB-regulated transcriptional coactivator 1 (CRTC1), was blocked in transfected neurons. Cell attach patch recordings combined with live multiphoton imaging demonstrated that the loss of CaNB1 did not prevent neurons from fully participating in electrographic seizure activity. Finally, dendrite reconstruction showed that the elimination of CaNB1 prevented seizure-induced decreases in both dendrite length and branch number. Results suggest that CaN plays a key role in seizure-induced dendrite growth suppression and may contribute to the neurobehavioral comorbidities of childhood epilepsy.
神经行为异常通常与难治性儿童癫痫有关。许多实验室的研究表明,经历过早期癫痫发作的大鼠和小鼠存在认知和社交障碍。然而,这些影响的细胞和分子机制尚不清楚。先前的实验表明,婴儿期反复发作的癫痫会同时抑制海马树突的生长,并损害学习和记忆。切片培养实验也证明了树突生长受到抑制。在这里,我们将钙调神经磷酸酶 B1(CaNB1) floxed 和 Thy1GFP-M 小鼠进行杂交,产生同时纯合 floxed CaNB1 和 Thy1GFP-M 转基因的小鼠。同窝出生的野生型 CaNB1 和 Thy1GFP-M 纯合小鼠作为对照。从这些小鼠的海马切片培养物中转染 AAV/hSyn-mCherry-Cre 病毒以从神经元中消除 CaNB1。免疫组织化学结果表明,CaNB1 至少从 90%的转染 CA1 锥体神经元中被消除。此外,钙调神经磷酸酶依赖性的 CREB 转录共激活因子 CREB 调节转录共激活因子 1(CRTC1)的核易位在转染神经元中被阻断。细胞附着斑记录与活多光子成像相结合的实验表明,CaNB1 的缺失并不能阻止神经元完全参与电生理癫痫活动。最后,树突重建表明,CaNB1 的消除阻止了癫痫引起的树突长度和分支数量的减少。结果表明,钙调神经磷酸酶在癫痫诱导的树突生长抑制中起关键作用,并可能导致儿童癫痫的神经行为共病。