Kang P B, Krishnamoorthy K S, Jones R M, Shapiro F D, Darras B T
Department of Neurology, Children's Hospital Boston and Harvard Medical School, Fegan 11, 300 Longwood Avenue, Boston, MA 02115, USA.
Neuromuscul Disord. 2006 Aug;16(8):492-4. doi: 10.1016/j.nmd.2006.05.004. Epub 2006 Jun 22.
Spinal muscular atrophy type III (SMA III, Kugelberg-Welander disease) typically presents with symmetric proximal weakness, areflexia, and hypotonia. We present four children with spinal muscular atrophy type III who had atypical phenotypes. Three patients clearly had asymmetric weakness at presentation and two had upper motor neuron signs in the lower extremities (one patient had both features). Two of the patients had prolonged evaluations before the diagnosis was made. All patients had Gowers signs and two had pes planus. In patients with proximal muscle weakness the presence of asymmetrical weakness, upper motor neuron signs, or both, may be compatible with spinal muscular atrophy type III. The diagnosis of spinal muscular atrophy should be considered when other possibilities have been excluded.
III型脊髓性肌萎缩症(SMA III,库格尔贝格-韦兰德病)通常表现为对称性近端肌无力、反射消失和肌张力减退。我们报告了4例具有非典型表型的III型脊髓性肌萎缩症患儿。3例患者在就诊时明显存在不对称肌无力,2例下肢有上运动神经元体征(1例患者兼具这两种特征)。其中2例患者在确诊前接受了长时间评估。所有患者均有戈氏征,2例有扁平足。对于近端肌无力患者,存在不对称肌无力、上运动神经元体征或两者兼具,可能符合III型脊髓性肌萎缩症。当排除其他可能性时,应考虑脊髓性肌萎缩症的诊断。