Merlini L, Granata C, Ballestrazzi A, Marini M L
Muscle Clinic, Istituto Ortopedico Rizzoli, Bologna, Italy.
J Child Neurol. 1989 Oct;4(4):274-82. doi: 10.1177/088307388900400405.
We studied eight patients with rigid spine syndrome aged 8 to 20 years at the time of first examination. Muscle weakness, rigid spine, and flexion contracture of elbows and ankles were noted in the first 6 years of age. Radiological study of the cervical spine revealed considerable reduction not only of flexion, but also of extension, of the neck. The "alligator sign" was demonstrated. Progression of scoliosis and of contractures reduced the functional capacity in six patients. A restrictive ventilatory syndrome was observed in all, while central apneas with oxygen desaturation were shown in two. Cardiac arrhythmias were observed in four. We suggest that a distinction should be made between myopathic rigid spine and rigid spine syndrome. Myopathic rigid spine is a clinical sign which is rarely sought but is present in several types of well-defined myopathies, meaning the myopathic involvement of the extensor muscles of the spine. Rigid spine syndrome is an axial myopathy with peculiar clinical and radiographic signs and a characteristic natural history.
我们研究了8例初次检查时年龄在8至20岁的僵硬脊柱综合征患者。在6岁前即发现肌无力、脊柱僵硬以及肘和踝关节的屈曲挛缩。颈椎的放射学研究显示,不仅颈部前屈,而且后伸也明显受限。出现了“鳄鱼征”。6例患者的脊柱侧弯和挛缩进展导致功能能力下降。所有患者均观察到限制性通气综合征,2例出现伴有氧饱和度降低的中枢性呼吸暂停。4例观察到心律失常。我们建议应区分肌病性脊柱僵硬和僵硬脊柱综合征。肌病性脊柱僵硬是一种临床体征,很少被关注,但存在于几种明确的肌病类型中,指脊柱伸肌的肌病性受累。僵硬脊柱综合征是一种轴索性肌病,具有独特的临床和影像学表现以及特征性的自然病程。