New York University Abu Dhabi , PO Box 129188, Abu Dhabi , United Arab Emirates.
Department of Genetics , University of Georgia , Athens , Georgia 30602 , United States.
ACS Chem Neurosci. 2019 Jan 16;10(1):266-278. doi: 10.1021/acschemneuro.8b00231. Epub 2018 Sep 24.
γ-Amino butyric acid (GABA) mediated signaling is critical in the central and enteric nervous systems, pancreas, lungs, and other tissues. It is associated with many neurological disorders and craniofacial development. Glutamic acid decarboxylase (GAD) synthesizes GABA from glutamate, and knockdown of the gad1 gene results in craniofacial defects that are lethal in zebrafish. To bypass this and enable observation of the neurological defects resulting from knocking down gad1 expression, a photoactivatable morpholino oligonucleotide (MO) against gad1 was prepared by cyclization with a photocleavable linker rendering the MO inactive. The cyclized MO was stable in the dark and toward degradative enzymes and was completely linearized upon brief exposure to 405 nm light. In the course of investigating the function of the ccMOs in zebrafish, we discovered that zebrafish possess paralogous gad1 genes, gad1a and gad1b. A gad1b MO injected at the 1-4 cell stage caused severe morphological defects in head development, which could be bypassed, enabling the fish to develop normally, if the fish were injected with a photoactivatable, cyclized gad1b MO and grown in the dark. At 1 day post fertilization (dpf), light activation of the gad1b MO followed by observation at 3 and 7 dpf led to increased and abnormal electrophysiological brain activity compared to wild type animals. The photocleavable linker can be used to cyclize and inactivate any MO, and represents a general strategy to parse the function of developmentally important genes in a spatiotemporal manner.
γ-氨基丁酸(GABA)介导的信号转导在中枢和肠神经系统、胰腺、肺和其他组织中至关重要。它与许多神经紊乱和颅面发育有关。谷氨酸脱羧酶(GAD)将谷氨酸合成 GABA,gad1 基因的敲低导致斑马鱼出现颅面缺陷,这在斑马鱼中是致命的。为了克服这一点,并能够观察到敲低 gad1 表达所导致的神经缺陷,我们通过用光可裂解的连接子环化来制备针对 gad1 的光激活型吗啉代寡核苷酸(MO),从而使 MO 失活。在黑暗中,这种环化的 MO 对降解酶稳定且稳定,并且在短暂暴露于 405nm 光下时完全线性化。在研究 ccMO 在斑马鱼中的功能的过程中,我们发现斑马鱼拥有gad1 基因的同源基因,gad1a 和 gad1b。在 1-4 细胞期注射 gad1b MO 会导致头部发育的严重形态缺陷,如果将光激活型、环化的 gad1b MO 注射到鱼中并在黑暗中生长,则可以克服这些缺陷,使鱼正常发育。在受精后 1 天(dpf),光激活 gad1b MO 后,在 3 和 7 dpf 观察到的电生理脑活动比野生型动物增加且异常。光可裂解的连接子可用于环化和失活任何 MO,这代表了一种以时空方式解析发育重要基因功能的通用策略。