Pediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, South Dakota, United States of America.
Department of Pathology, University of Iowa Carver College of Medicine, Iowa City, Iowa, United States of America.
PLoS One. 2020 Feb 19;15(2):e0228222. doi: 10.1371/journal.pone.0228222. eCollection 2020.
Genetically modified swine disease models are becoming increasingly important for studying molecular, physiological and pathological characteristics of human disorders. Given the limited history of these model systems, there remains a great need for proven molecular reagents in swine tissue. Here, to provide a resource for neurological models of disease, we validated antibodies by immunohistochemistry for use in examining central nervous system (CNS) markers in a recently developed miniswine model of neurofibromatosis type 1 (NF1). NF1 is an autosomal dominant tumor predisposition disorder stemming from mutations in NF1, a gene that encodes the Ras-GTPase activating protein neurofibromin. Patients classically present with benign neurofibromas throughout their bodies and can also present with neurological associated symptoms such as chronic pain, cognitive impairment, and behavioral abnormalities. As validated antibodies for immunohistochemistry applications are particularly difficult to find for swine models of neurological disease, we present immunostaining validation of antibodies implicated in glial inflammation (CD68), oligodendrocyte development (NG2, O4 and Olig2), and neuron differentiation and neurotransmission (doublecortin, GAD67, and tyrosine hydroxylase) by examining cellular localization and brain region specificity. Additionally, we confirm the utility of anti-GFAP, anti-Iba1, and anti-MBP antibodies, previously validated in swine, by testing their immunoreactivity across multiple brain regions in mutant NF1 samples. These immunostaining protocols for CNS markers provide a useful resource to the scientific community, furthering the utility of genetically modified miniswine for translational and clinical applications.
基因修饰猪疾病模型在研究人类疾病的分子、生理和病理特征方面变得越来越重要。鉴于这些模型系统的历史有限,仍然需要在猪组织中使用经过验证的分子试剂。在这里,为了提供神经疾病模型的资源,我们通过免疫组织化学验证了抗体,用于检查最近开发的神经纤维瘤病 1 型(NF1)小型猪模型中的中枢神经系统(CNS)标志物。NF1 是一种常染色体显性遗传肿瘤易感性疾病,源于 NF1 基因突变,该基因编码 Ras-GTP 酶激活蛋白神经纤维瘤素。患者经典地在全身出现良性神经纤维瘤,并且还可能出现与神经相关的症状,如慢性疼痛、认知障碍和行为异常。由于针对神经疾病的猪模型,特别难以找到用于免疫组织化学应用的验证抗体,因此我们通过检查细胞定位和大脑区域特异性,对涉及神经胶质炎症(CD68)、少突胶质细胞发育(NG2、O4 和 Olig2)和神经元分化和神经传递(双皮质蛋白、GAD67 和酪氨酸羟化酶)的抗体进行免疫染色验证。此外,我们通过测试突变 NF1 样本中多个脑区的免疫反应性,证实了之前在猪中验证过的抗 GFAP、抗 Iba1 和抗 MBP 抗体的有用性。这些 CNS 标志物的免疫染色方案为科学界提供了有用的资源,进一步提高了基因修饰小型猪在转化和临床应用中的效用。