McEntagart M, Dunstan M, Bell C, Boltshauser E, Donaghy M, Harper P S, Williams N, Teare M D, Rahman N
Institute of Medical Genetics, University Hospital of Wales, Cardiff, UK.
J Neurol Neurosurg Psychiatry. 2002 Dec;73(6):762-5. doi: 10.1136/jnnp.73.6.762.
The peroneal muscular atrophy syndrome is the most common inherited disorder of the peripheral nervous system and has extensive clinical and genetic heterogeneity. Cranial nerve involvement is rare, though there are distinct peroneal muscular atrophy syndromes in which vocal cord paralysis is a characteristic feature. Among these dHMN-VII and HMSN-IIC are clinically similar but are differentiated by sensory involvement in HMSN-IIC. The gene for dHMN-VII, designated DHMNVP, has been localised to chromosome 2q14, but the location of the gene for HMSN-IIC is currently unknown. It has been suggested that dHMN-VII and HMSN II-C are allelic disorders.
To assess the contribution of the dHMN-VII predisposition gene to peroneal muscular atrophy syndromes associated with vocal cord weakness.
Linkage analysis of microsatellite markers at chromosome 2q14 was undertaken on two families, one affected by HMSN-IIC and a second manifesting vocal cord paralysis and sensorineural deafness in addition to distal muscular atrophy.
Two-point LOD scores at chromosome 2q14 markers encompassing the DHMNVP gene were negative in both families.
These results suggest that at least one further gene predisposing to distal muscular weakness in association with vocal cord paralysis is likely to exist, and that dHMN-VII and HMSN-IIC are unlikely to be allelic disorders. Analyses of further HMSN-IIC families are required to confirm this.
腓骨肌萎缩综合征是周围神经系统最常见的遗传性疾病,具有广泛的临床和遗传异质性。虽然存在明显的腓骨肌萎缩综合征,其中声带麻痹是一个特征性表现,但颅神经受累情况罕见。在这些综合征中,远端遗传性运动神经病VII型(dHMN-VII)和遗传性感觉运动神经病IIC型(HMSN-IIC)在临床上相似,但通过HMSN-IIC中的感觉受累情况进行区分。dHMN-VII的基因,命名为DHMNVP,已定位到2号染色体长臂14区,但HMSN-IIC的基因位置目前尚不清楚。有人提出dHMN-VII和HMSN II-C是等位基因疾病。
评估dHMN-VII易感基因对与声带无力相关的腓骨肌萎缩综合征的影响。
对两个家系进行了2号染色体长臂14区微卫星标记的连锁分析,一个家系患有HMSN-IIC,另一个家系除了远端肌肉萎缩外,还表现为声带麻痹和感音神经性耳聋。
在包含DHMNVP基因的2号染色体长臂14区标记处,两个家系的两点连锁对数得分均为阴性。
这些结果表明,可能至少存在一个与声带麻痹相关的导致远端肌肉无力的易感基因,并且dHMN-VII和HMSN-IIC不太可能是等位基因疾病。需要对更多的HMSN-IIC家系进行分析以证实这一点。