Lee Choong H, O'Connor Amber K, Yang Chaozhe, Tate Joshua M, Schoeb Trenton R, Flint Jeremy J, Blackband Stephen J, Guay-Woodford Lisa M
Department of Neuroscience, University of Florida, Gainesville, Florida McKnight Brain Institute, University of Florida, Gainesville, Florida.
Center for Translational Science, Children's National Health System, Washington, District of Columbia.
Physiol Rep. 2015 Aug;3(8). doi: 10.14814/phy2.12517.
Polycystic kidney disease (PKD) is transmitted as either an autosomal dominant or recessive trait and is a major cause of renal failure and liver fibrosis. The cpk mouse model of autosomal recessive PKD (ARPKD) has been extensively characterized using standard histopathological techniques after euthanasia. In the current study, we sought to validate magnetic resonance microscopy (MRM) as a robust tool for assessing the ARPKD phenotype. We used MRM to evaluate the liver and kidney of wild-type and cpk animals at resolutions <100 μm and generated three-dimensional (3D) renderings for pathological evaluation. Our study demonstrates that MRM is an excellent method for evaluating the complex, 3D structural defects in this ARPKD mouse model. We found that MRM was equivalent to water displacement in assessing kidney volume. Additionally, using MRM we demonstrated for the first time that the cpk liver exhibits less extensive ductal arborization, that it was reduced in volume, and that the ductal volume was disproportionately smaller. Histopathology indicates that this is a consequence of bile duct malformation. With its reduced processing time, volumetric information, and 3D capabilities, MRM will be a useful tool for future in vivo and longitudinal studies of disease progression in ARPKD. In addition, MRM will provide a unique tool to determine whether the human disease shares the newly appreciated features of the murine biliary phenotype.
多囊肾病(PKD)以常染色体显性或隐性性状遗传,是肾衰竭和肝纤维化的主要原因。常染色体隐性多囊肾病(ARPKD)的cpk小鼠模型在安乐死后已使用标准组织病理学技术进行了广泛的特征描述。在本研究中,我们试图验证磁共振显微镜(MRM)作为评估ARPKD表型的可靠工具。我们使用MRM在分辨率小于100μm的情况下评估野生型和cpk动物的肝脏和肾脏,并生成三维(3D)渲染图用于病理学评估。我们的研究表明,MRM是评估该ARPKD小鼠模型中复杂的3D结构缺陷的极佳方法。我们发现,MRM在评估肾脏体积方面与水置换法相当。此外,使用MRM我们首次证明cpk肝脏的导管分支较少,体积减小,且导管体积不成比例地更小。组织病理学表明这是胆管畸形的结果。由于其处理时间缩短、具有体积信息和3D功能,MRM将成为未来对ARPKD疾病进展进行体内和纵向研究的有用工具。此外,MRM将提供一个独特的工具来确定人类疾病是否具有新发现的小鼠胆汁表型特征。