Ohto T, Iwasaki N, Fujiwara J, Ohkoshi N, Kimura S, Kawade K, Tanaka R, Matsui A
Department of Pediatrics, Institute of Clinical Medicine, University of Tsukuba, Tsukuba, Ibaraki, Japan.
Neuropediatrics. 2004 Oct;35(5):274-8. doi: 10.1055/s-2004-821254.
We report on a 10-year-old girl with anhidrosis and insensibility to pain, but no severe mental retardation or self-mutilation, diagnosed as hereditary sensory and autonomic neuropathy type IV (HSAN IV). Genetic analysis of her TRKA gene, which is responsible for HSAN IV, revealed two novel missense mutations in the tyrosine kinase domain. Cardiovascular autonomic nervous system function tests showed normal muscle sympathetic nerve activity associated with arterial baroreflex, reduced skin sympathetic nerve activity in the second and fifth fingers and palms, and abnormal circadian rhythm of cardiovascular autonomic nervous system. These findings have never before been reported in HSAN IV and may provide a clue to the neurological pathophysiology of this disease.
我们报告了一名10岁女孩,她患有无汗症且对疼痛不敏感,但无严重智力发育迟缓或自残行为,被诊断为遗传性感觉和自主神经病变IV型(HSAN IV)。对其负责HSAN IV的TRKA基因进行遗传分析,发现在酪氨酸激酶结构域有两个新的错义突变。心血管自主神经系统功能测试显示,与动脉压力反射相关的肌肉交感神经活动正常,第二和第五手指及手掌的皮肤交感神经活动减少,以及心血管自主神经系统的昼夜节律异常。这些发现此前从未在HSAN IV中报道过,可能为该疾病的神经病理生理学提供线索。