Chance P F, Reilly M
Division of Neurology Research, Children's Hospital of Philadelphia, PA 19104.
Curr Opin Neurol. 1994 Oct;7(5):372-80. doi: 10.1097/00019052-199410000-00002.
Charcot-Marie-Tooth neuropathy (CMT) type 1 is a genetically heterogeneous group of chronic demyelinating polyneuropathies with loci mapping to chromosome 17 (CMT1A), chromosome 1 (CMT1B), the X chromosome (CMTX), and to another unknown autosome (CMT1C). CMT1A is most often associated with a tandem 1.5-Mb duplication in chromosome 17p11.2-12, or in rare patients may result from a point mutation in the peripheral myelin protein-22 (PMP22) gene. CMT1B is associated with point mutations in the myelin protein zero (P0) gene. The molecular defect in CMT1C is unknown. CMTX is associated with mutations in the connexin 32 gene. CMT2 is an axonal neuropathy of undetermined cause. One form of CMT2 maps to chromosome 1p36 (CMT2A). Dejerine-Sottas disease is a severe, infantile-onset demyelinating polyneuropathy that may be associated with point mutations in either the PMP22 gene or the P0 gene. Hereditary neuropathy with liability to pressure palsies (HNPP) is a recurrent, episodic demyelinating neuropathy. HNPP is associated with a 1.5-Mb deletion in chromosome 17p11.2-12 and may result from reduced expression of the PMP22 gene. Most examples of CMT1A and HNPP are reciprocal duplication or deletion syndromes originating from unequal crossover during germ cell meiosis. Familial amyloid polyneuropathy (FAP) is an autosomal dominant disorder that classically presents with a sensory peripheral neuropathy and early autonomic involvement. Transthyretin (TTR) is the most common constituent amyloid fibril protein deposited in FAP, and there are now 28 point mutations in the TTR gene described in TTR-related FAP. Liver transplantation looks promising as a treatment for TTR-related FAP.
1型夏科-马里-图思神经病(CMT)是一组具有遗传异质性的慢性脱髓鞘性多发性神经病,其基因座分别定位于17号染色体(CMT1A)、1号染色体(CMT1B)、X染色体(CMTX)以及另一条未知常染色体(CMT1C)。CMT1A最常与17p11.2 - 12染色体上1.5Mb的串联重复相关,或在罕见患者中可能由外周髓鞘蛋白22(PMP22)基因突变引起。CMT1B与髓鞘蛋白零(P0)基因突变相关。CMT1C的分子缺陷尚不清楚。CMTX与连接蛋白32基因突变相关。CMT2是一种病因不明的轴索性神经病。CMT2的一种形式定位于1p36染色体(CMT2A)。德热里纳-索塔斯病是一种严重的婴儿期起病的脱髓鞘性多发性神经病,可能与PMP22基因或P0基因的点突变有关。遗传性压力易感性神经病(HNPP)是一种复发性、发作性脱髓鞘性神经病。HNPP与17p11.2 - 12染色体上1.5Mb的缺失相关,可能由PMP22基因表达降低所致。CMT1A和HNPP的大多数病例是源于生殖细胞减数分裂期间不等交换的相互重复或缺失综合征。家族性淀粉样多发性神经病(FAP)是一种常染色体显性疾病,典型表现为感觉性周围神经病并早期累及自主神经。转甲状腺素蛋白(TTR)是FAP中沉积的最常见的淀粉样原纤维蛋白成分,目前在与TTR相关的FAP中已描述了TTR基因的28个点突变。肝移植作为治疗与TTR相关的FAP的方法看起来很有前景。