Hirose Takuya, Suzuki Ippei, Takahashi Naoki, Fukada Toshiyuki, Tangkawattana Prasarn, Takehana Kazushige
Laboratory of Microanatomy, School of Veterinary Medicine, Rakuno Gakuen University, Ebetsu, Hokkaido 069-8501, Japan.
Molecular and Cellular Physiology, Faculty of Pharmaceutical Sciences, Tokushima Bunri University, Tokushima 770-8055, Japan.
J Vet Med Sci. 2018 May 18;80(5):814-818. doi: 10.1292/jvms.18-0019. Epub 2018 Mar 22.
Ehlers-Danlos syndrome (EDS) is a group of hereditary diseases caused by mutation of extracellular matrix-related genes. Recently, spondylodysplastic EDS-Zip13 (spEDS-Zip13: OMIM 612350) was recognized as a new EDS type. This current study could reveal various morphometric differences of collagenous population in the proper substance of cornea between the wild type and spEDS-Zip13-knockout (Zip13-KO) mice. Blockade of Smad-signaling pathway might initiate these alterations. Predilected dissimilarity in level of transcriptional activity probably dictated morphology of keratocyte and shape and electron density of its nucleus. In addition, the imbalance of proteoglycans and glycosaminoglycans would also affect the diameter and arrangement of collagen fibrils. These findings would be considered as vulnerable characteristics of corneal stroma of the Zip13-KO mice.
埃勒斯-当洛综合征(EDS)是一组由细胞外基质相关基因突变引起的遗传性疾病。最近,脊柱发育不良性EDS-Zip13(spEDS-Zip13:OMIM 612350)被确认为一种新的EDS类型。当前这项研究能够揭示野生型和spEDS-Zip13基因敲除(Zip13-KO)小鼠角膜固有层中胶原群体的各种形态计量学差异。Smad信号通路的阻断可能引发了这些改变。转录活性水平上的偏好性差异可能决定了角膜细胞的形态及其细胞核的形状和电子密度。此外,蛋白聚糖和糖胺聚糖的失衡也会影响胶原纤维的直径和排列。这些发现将被视为Zip13-KO小鼠角膜基质的易损特征。