Parnes S, Karpati G, Carpenter S, Kin N M, Wolfe L S, Suranyi L
Arch Neurol. 1985 Dec;42(12):1176-80. doi: 10.1001/archneur.1985.04060110058016.
Three patients from two families had an unusual phenotypical variant of late-onset hexosaminidase-A deficiency. The clinical picture was dominated by spinal motor neuron involvement mimicking juvenile-onset spinal muscular atrophy. Atypical features included prominent muscle cramps, postural and action tremor, recurrent psychosis, incoordination, corticospinal and corticobulbar involvement, and dysarthria. The presence of these atypical features in patients whose lower motor neuron involvement would otherwise be consistent with juvenile-onset spinal muscular atrophy should raise the suspicion of the presence of hexosaminidase-A deficiency and GM2 gangliosidosis that can be proved by appropriate enzyme assays.
来自两个家族的三名患者患有迟发性己糖胺酶 A 缺乏症的一种不寻常表型变异。临床表现以模仿青少年型脊髓性肌萎缩症的脊髓运动神经元受累为主。非典型特征包括明显的肌肉痉挛、姿势性和动作性震颤、反复出现的精神病、共济失调、皮质脊髓束和皮质延髓束受累以及构音障碍。在那些下运动神经元受累情况原本与青少年型脊髓性肌萎缩症相符的患者中出现这些非典型特征,应引起对己糖胺酶 A 缺乏症和 GM2 神经节苷脂贮积症的怀疑,这可通过适当的酶测定来证实。