Jankovic J, Kirkpatrick J B, Blomquist K A, Langlais P J, Bird E D
Neurology. 1985 Feb;35(2):227-34. doi: 10.1212/wnl.35.2.227.
We studied a 68-year-old man who died after 13 years of progressive dementia, rigidity, bradykinesia, mild tremor, stooped posture, slow and shuffling gait, dystonia, blepharospasm, apraxia of eyelid opening, anarthria, aphonia, and incontinence. At autopsy, he had generalized brain atrophy with large deposits of iron pigment in the globus pallidus, caudate, and substantia nigra. Axonal spheroids were found in the globus pallidus, substantia nigra, medulla, and spinal cord. The neurochemical analysis of the brain revealed marked loss of dopamine in the nigral-striatal areas, with relative preservation of dopamine in the limbic areas. This is the oldest case of familial Hallervorden-Spatz disease reported and the first with neurochemical analysis of the brain.
我们研究了一名68岁男性,他在出现进行性痴呆、僵硬、运动迟缓、轻度震颤、弯腰姿势、缓慢拖曳步态、肌张力障碍、眼睑痉挛、眼睑开合失用、构音障碍、失音和尿失禁13年后死亡。尸检时,他出现广泛性脑萎缩,苍白球、尾状核和黑质有大量铁色素沉积。在苍白球、黑质、延髓和脊髓中发现了轴突球体。对大脑的神经化学分析显示,黑质纹状体区域多巴胺明显缺失,而边缘系统区域多巴胺相对保留。这是报道的家族性Hallervorden-Spatz病最年长的病例,也是首例对大脑进行神经化学分析的病例。