Hitoshi Hayashida, Yukimasa Arita, Kishin Koh, Yoshihisa Takiyama, Koji Ikezoe
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引用次数: 0
摘要
一名 52 岁的男子自孩提时代起就患有听力损失,四十多岁时还患上了反复发作的足部溃疡和骨髓炎。他出现步态障碍和构音障碍,并分别在四年和一个月后加重。神经系统检查显示,他有认知障碍、下肢近端无力、全身反射亢进、共济失调、以深感觉为主的感觉障碍、尿潴留和步态不稳。在神经传导检查中,上下肢均未诱发感觉神经动作电位。由于他的祖母也有类似症状,我们对他进行了基因分析,结果发现 DNA 甲基转移酶 1 基因有一个错义突变(c.1483T>C,p.Y495H)。随后,他被诊断为遗传性感觉和自主神经病变 1E(HSAN1E)。值得注意的是,在 HSAN1E 中可以观察到深腱反射增强。
[Hereditary sensory and autonomic neuropathy 1E showing hyperreflexia: a case report].
A 52-year-old man had developed hearing loss since childhood, as well as recurrent foot ulcers and osteomyelitis since his forties. He presented with gait disturbance and dysarthria that had worsened over four years and a month, respectively. Neurological exams revealed cognitive impairment, proximal weakness of the lower extremities, generalized hyperrflexia, ataxia, sensory disturbances predominant in deep sensation, urinary retention, and gait instability. On nerve conduction study, no sensory nerve action potentials were evoked in the upper and lower limbs. Since his grandmother suffered from similar symptoms, we investigated genetic analysis, which revealed a missense mutation (c.1483T>C, p.Y495H) in DNA methyltransferase 1 gene. He was subsequently diagnosed with hereditary sensory and autonomic neuropathy 1E (HSAN1E). It is important to recognize that increased deep tendon reflex can be observed in HSAN1E.