Laboratory of Anatomy, 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.
Cell Tissue Res. 2019 Apr;376(1):137-141. doi: 10.1007/s00441-018-2977-9. Epub 2019 Jan 4.
Ehlers-Danlos syndrome (EDS) is a collection of inheritable diseases involving the musculoskeletal, integumentary and visual systems. Spondylodysplastic EDS-ZIP13 (spEDS-ZIP13: OMIM 612350) was recently defined as a new form of EDS. Although vasculitis has been found in many spEDS-ZIP13 patients, vascular pathology has not been included as a pathognomonic lesion of this type of EDS. We investigate the morphometry of the thoracic aorta in wild-type and Zip13-knockout (Zip13-KO) mice. Our assessment found abnormalities in the number and morphology of elastic and cellular components in the aortic wall, especially the tunica media, of Zip13-KO mice, indicating aortic fragility. Accordingly, our major findings (vascular smooth muscle cells with small nuclei, small percentage of elastic membrane area per tunica media, many large elastic flaps) should be considered vulnerable characteristics indicating fragility of the aorta in patients with spEDS-ZIP13.
埃勒斯-当洛斯综合征(EDS)是一组涉及肌肉骨骼、皮肤和视觉系统的遗传性疾病。最近将脊柱发育不良型 EDS-ZIP13(spEDS-ZIP13:OMIM 612350)定义为 EDS 的一种新形式。尽管许多 spEDS-ZIP13 患者中发现了血管炎,但血管病理学尚未被纳入这种类型 EDS 的特征性病变。我们研究了野生型和 Zip13 敲除(Zip13-KO)小鼠的胸主动脉形态计量学。我们的评估发现,Zip13-KO 小鼠的主动脉壁弹性和细胞成分的数量和形态存在异常,尤其是中膜,这表明主动脉脆弱。因此,我们的主要发现(具有小核的血管平滑肌细胞、中膜每单位弹性膜面积的小百分比、许多大的弹性瓣)应被视为脆弱特征,表明 spEDS-ZIP13 患者的主动脉脆弱。