Mandich P, James R, Nassani S, Defferrari R, Bellone E, Mancardi G, Schenone A, Abbruzzese M, Rocchi M, Ajmar F
Institute of Biology and Genetics, Genoa, Italy.
J Neurol. 1995 May;242(5):295-8. doi: 10.1007/BF00878871.
Hereditary neuropathy with a liability to pressure palsies (HNPP) is an autosomal dominant disorder characterized by recurrent pressure palsies generally precipitated by minor trauma; weakness and paraesthesia usually improve and recover completely in a few months. By Southern blotting and fluorescent in situ hybridization analysis we confirm the presence of a 17p11.2 deletion in familial and in isolated cases of HNPP, suggesting that molecular analysis of the 17p11.2 region could also be a reliable and non-invasive method of diagnosis in sporadic cases, where a correct diagnosis usually requires a nerve biopsy. Although HNPP is a mild disease and not all patients seek medical attention, a presymptomatic diagnosis is useful for assessing the risk during genetic counselling, due to the inheritance of the mutation.
遗传性压力易感性周围神经病(HNPP)是一种常染色体显性疾病,其特征为反复出现的压迫性麻痹,通常由轻微创伤诱发;肌无力和感觉异常通常在数月内改善并完全恢复。通过Southern印迹法和荧光原位杂交分析,我们证实了家族性和散发性HNPP病例中存在17p11.2缺失,这表明对17p11.2区域进行分子分析也可能是散发性病例中一种可靠且非侵入性的诊断方法,而在散发性病例中,通常需要进行神经活检才能做出正确诊断。尽管HNPP是一种轻症疾病,并非所有患者都会就医,但由于突变具有遗传性,症状前诊断对于在遗传咨询期间评估风险是有用的。