Mitani M, Jinnai K, Takahashi K, Koide R, Tsuji S
Department of Neurology, National Sanatorium Hyogo-Chuo Hospital.
Rinsho Shinkeigaku. 2000 Jun;40(6):600-4.
We report a case of NARP with a T-to-C point mutation at nt 8993 of mitochondrial DNA. A 37-year old man with mild mental retardation, retinitis pigmentosa, and clonic-tonic seizure was admitted to our hospital. The neurological examination revealed scanning speech, dystonic neck turning to the left side, and pyramidal tract signs. Serum-, CSF-lactate and pyruvate level were slightly elevated. Brain MRI findings showed cerebral atrophy, cerebellar cortical atrophy accompanied with dilation of forth ventricle, and high intensity lesions in the bilateral lenticular nuclei on T2 weighted images. Nucleotide sequence analysis of the mitochondrial DNA in the leukocytes demonstrated a T-to-C point mutation at nt 8993. To our knowledge, this is the first report of a Japanese patient with NARP associated with the T-to-C mutation at nt 8993 of mt DNA. Mitochondrial DNA analysis should be considered in the differential diagnosis of patients with retinitis pigmentosa and various neurological signs.
我们报告了一例线粒体DNA第8993位核苷酸发生T到C点突变的神经病变、共济失调和色素性视网膜炎(NARP)病例。一名患有轻度智力障碍、色素性视网膜炎和阵挛 - 强直发作的37岁男性入住我院。神经系统检查发现断续言语、颈部向左扭转的肌张力障碍以及锥体束征。血清、脑脊液乳酸和丙酮酸水平略有升高。脑部MRI检查结果显示脑萎缩、小脑皮质萎缩伴第四脑室扩张,以及T2加权图像上双侧豆状核的高强度病变。白细胞中线粒体DNA的核苷酸序列分析显示第8993位核苷酸发生T到C点突变。据我们所知,这是日本首例与线粒体DNA第8993位T到C突变相关的NARP患者报告。对于患有色素性视网膜炎和各种神经体征的患者,在鉴别诊断时应考虑线粒体DNA分析。