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致密性骨发育不全罕见病例:长骨骨折处理中的技术难题

A rare case of pycnodysostosis: Technical difficulties in managing long bone fractures.

作者信息

Romans M, Sambandam Balaji, Moses Justin, Pragash M

机构信息

Dept of Orthopedics, Sri Manakula Vinayagar Medical College and Hospital, Puducherry, India.

出版信息

J Clin Orthop Trauma. 2020 Mar-Apr;11(2):332-338. doi: 10.1016/j.jcot.2018.09.012. Epub 2018 Sep 26.

Abstract

Pycnodysostosis is a rare inherited disorder of autosomal recessive trait causing cathepsin K deficiency, leading to failure of osteoclastic activity. Brittle and sclerotic bones which are prone for frequent fractures is the characteristic feature of this congenital disorder. Despite good healing potential there are few issues in the management of fractures in pycnodysostosis patients. In this article we report the challenges faced in managing a fracture of the femoral shaft in a 12 year old girl with pycnodysostosis. For early rehabilitation and to avoid deformity and shortening, we opted for surgical fixation over conservative treatment. Narrow medullary canal ruled out the option for titanium elastic nail fixation. 4.5mm dynamic compression plate was used to fix the fracture. Sclerotic bone made drilling extremely difficult. Deformed femoral shaft allowed plating over the anterior surface only, instead of the routine lateral surface plating. Postoperative fracture healing was satisfactory. Implant was removed after 18 months.

摘要

致密性骨发育不全是一种罕见的常染色体隐性遗传疾病,可导致组织蛋白酶K缺乏,进而引起破骨细胞活性丧失。骨骼脆弱且硬化,易于频繁骨折,是这种先天性疾病的特征。尽管骨折愈合潜力良好,但致密性骨发育不全患者骨折的处理仍存在一些问题。在本文中,我们报告了一名12岁致密性骨发育不全女童股骨干骨折处理中所面临的挑战。为了早期康复并避免畸形和缩短,我们选择手术固定而非保守治疗。狭窄的髓腔排除了使用钛弹性髓内钉固定的选择。使用4.5mm动力加压钢板固定骨折。硬化骨使得钻孔极为困难。股骨干变形使得只能在前侧而非常规的外侧进行钢板固定。术后骨折愈合情况令人满意。18个月后取出植入物。

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Clinical and radiographic features of pycnodysostosis: A case report.致密性骨发育不全的临床和影像学特征:一例报告。
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