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PMID: 25337104 已发表 · ppublish 英语

Rapid genetic screening of Charcot-Marie-Tooth disease type 1A and hereditary neuropathy with liability to pressure palsies patients.

Neural regeneration research ·第 7 卷 ·第 32 期 ·2014-10-22

Li Xiaobo, Zi Xiaohong, Li Lin, Zhan Yajing, Huang Shunxiang, Li Jin, Li Xuning, Li Xigui, Hu Zhengmao, Xia Kun, Tang Beisha, Zhang Ruxu

摘要

We used the allele-specific PCR-double digestion method on peripheral myelin protein 22 (PMP22) to determine duplication and deletion mutations in the proband and family members of one family with Charcot-Marie-Tooth disease type 1 and one family with hereditary neuropathy with liability to pressure palsies. The proband and one subclinical family member from the Charcot-Marie-Tooth disease type 1 family had a PMP22 gene duplication; one patient from the hereditary neuropathy with liability to pressure palsies family had a PMP22 gene deletion. Electron microscopic analysis of ultrathin sections of the superficial peroneal nerve from the two probands demonstrated demyelination and myelin sheath hyperplasia, as well as an 'onion-like' structure in the Charcot-Marie-Tooth disease type 1A patient. We observed an irregular thickened myelin sheath and 'mouse-nibbled'-like changes in the patient with hereditary neuropathy with liability to pressure palsies. In the Charcot-Marie-Tooth disease type 1A patient, nerve electrophysiological examination revealed moderate-to-severe reductions in the motor and sensory conduction velocities of the bilateral median nerve, ulnar nerve, tibial nerve, and sural nerve. Moreover, the compound muscle action potential amplitude was decreased. In the patient with hereditary neuropathy with liability to pressure palsies, the nerve conduction velocity of the bilateral tibial nerve and sural nerve was moderately reduced, and the nerve conduction velocity of the median nerve and ulnar nerve of both upper extremities was slightly reduced.

关键词
Charcot-Marie-Tooth disease PCR-double digestion method action potential gene mutation hereditary neuropathy with liability to pressure palsies myelin sheath neural regeneration neuropathology peripheral myelin protein 22
文献信息
期刊
Neural regeneration research
期刊简称
Neural Regen Res
发表日期
2014-10-22
收录日期
2014-10-22
更新日期
2014-10-25
语言
英语
国家/地区
India
NLM ID
101316351
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