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Development of spinal deformities in the tight-skin mouse

机译:紧肤小鼠脊柱畸形的发展

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摘要

Tight-skin (TSK) mice are commonly used as an animal model to study the pathogenesis of Marfan syndrome (MFS), but little is known of their skeletal phenotype and in particular of the development of the spinal deformities, common in MFS. Here we examined growth of the axial skeletons of TSK and wild-type(B6) mice during their period of rapid growth. The whole bodies of mice, 4–12 weeks of age, were scanned after sacrifice, by micro-computed tomography (microCT). We reconstructed three-dimensional models of the spine and ribs, and measured vertebral body heights and rib lengths using the Mac-based image-processing software “OsiriX”. Although the TSK mice were smaller than the B6 mice at 4 weeks, they experienced an early growth spurt and by 8 weeks the height, but not the width, of the vertebral body was significantly greater in the TSK mice than the B6 mice. Measurement of the angles of scoliotic and kyphotic curves post-mortem in the mice was problematic, hence we measured changes that develop in skeletal elements in these disorders. As a marker of kyphosis, we measured anterior wedging of the vertebral bodies; as a marker for scoliosis we measured asymmetries in rib length. We found, unlike in the B6 mice where the pattern was diffuse, wedging in TSK mice was directly related to spinal level and peaked steeply at the thoracolumbar junction. There was also significant asymmetry in length of the ribs in the TSK mice, but not in the B6 mice. The TSK mice thus appear to exhibit spinal deformities seen in MFS and could be a useful model for gaining understanding of the mechanisms of development of scoliosis and kyphosis in this disorder.
机译:紧密皮肤(TSK)小鼠通常用作研究马凡综合症(MFS)发病机理的动物模型,但对其骨骼表型,尤其是MFS中常见的脊柱畸形的发展知之甚少。在这里,我们检查了TSK和野生型(B6)小鼠快速生长期间其轴向骨骼的生长。处死后,通过微型计算机断层扫描(microCT)对4至12周龄的小鼠的整个身体进行扫描。我们使用基于Mac的图像处理软件“ OsiriX”重建了脊柱和肋骨的三维模型,并测量了椎体高度和肋骨长度。尽管TSK小鼠在4周时比B6小鼠小,但它们经历了早期生长突增,TSK小鼠中椎体的高度(而不是宽度)增加了8周,比B6小鼠大得多。在小鼠中测量死后脊柱侧弯和后凸曲线的角度是有问题的,因此我们测量了这些疾病中骨骼元素发生的变化。作为脊柱后凸的标志,我们测量了椎体的前楔形。作为脊柱侧弯的标志,我们测量了肋骨长度的不对称性。我们发现,与B6小鼠中的模式分散的情况不同,TSK小鼠中的楔形与脊髓水平直接相关,并在胸腰交界处陡峭地达到峰值。在TSK小鼠中,但在B6小鼠中,肋骨的长度也存在明显的不对称性。因此,TSK小鼠似乎表现出在MFS中看到的脊柱畸形,并且可能是一种有用的模型,可用于了解这种疾病的脊柱侧凸和后凸畸形的发展机制。

著录项

  • 期刊名称 Bone Research
  • 作者

    Bing Li; Jill PG Urban; Jing Yu;

  • 作者单位
  • 年(卷),期 2017(5),-1
  • 年度 2017
  • 页码 16053
  • 总页数 9
  • 原文格式 PDF
  • 正文语种
  • 中图分类 R6;
  • 关键词

  • 入库时间 2022-08-17 13:54:29

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