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A Severe Progressive Kyphoscoliosis Secondary to Arthrogryposis Multiplex Congenita. Case Report Yoji Ogura 1 , Kota Watanabe 2 , Takashi Tsuji 1 , Ken Ishii 1 , Hironari Takaishi 1 , Masaya Nakamura 1 , Yoshiaki Toyama 1 , Kazuhiro Chiba 1 , Morio Matsumoto 1 1Department of Orthopaedic Surgery, Keio University 2Department of Advanced Therapy for Spine and Spinal Cord Diseases, Keio University Keyword: 先天性多発性関節拘縮症 , arthrogryposis multiplex congenita , 後側弯症 , kyphoscoliosis , 心肺停止 , cardiopulmonary arrest pp.855-860
Published Date 2009/8/25
DOI https://doi.org/10.11477/mf.1408101577
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 We report a case of arthrogryposis multiplex congenita (AMC) complicated by severe progressive kyphoscoliosis. A 14-year-old girl with AMC presented with a severe progressive trunk deformity and was referred to our clinic. Radiography revealed 96° scoliosisand 58° kyphosis in the thoracic spine. The patient also exhibited severe restrictive pulmonary dysfunction (%VC:18%). We attempted halo-gravity traction prior to corrective surgery. During the halo ring attachment, cardiopulmonary arrest occurred twice because of difficulty in intubation, but resuscitation was successful. Following halo traction for six weeks, posterior correction surgery with a pedicle-screw-only construct was performed, but the extensive rigid curvature and fragility of the vertebrae hampered optimal correction of the deformity. Since the scoliosis in AMC patients is often extremely rigid and progressive, early surgical intervention should be considered, and meticulous preoperative evaluation of both pulmonary function and the feasibility of intubation is mandatory.


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電子版ISSN 1882-1286 印刷版ISSN 0557-0433 医学書院

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