Tyson J, Malcolm S, Thomas P K, Harding A E
Molecular Genetics Unit, Institute of Child Health, London, U.K.
Ann Neurol. 1996 Feb;39(2):180-6. doi: 10.1002/ana.410390207.
We investigated 51 patients with multifocal neuropathies for the deletion of chromosome 17p11.2 described in families with hereditary neuropathy with liability to pressure palsies (HNPP). The deletion was detected in 24 patients, including 19 patients from 14 of 15 families in whom HNPP had been considered likely on clinical, neurophysiological, and/or pathological grounds. One patient with a deletion had rather unusual clinical features for HNPP, presenting with a progressive scapuloperoneal syndrome. Overall, 7 (37%) of the 19 index patients with the deletion had no affected relatives, and less than half had evidence of a generalized neuropathy on examination. Peripheral nerve lesions were related to pressure in only 15 (62%) of the patients with the deletion. Nerve conduction studies in 23 of 25 patients and relatives studied showed a fairly uniform pattern of moderate prolongation of distal sensory and motor latencies and slowing of conduction velocities, and variable reduction of sensory or evoked muscle action potential amplitudes. The patients investigated who did not have a deletion of 17p11.2 were heterogeneous and included those with recurrent and/or familial neuralgic amyotrophy, two or more peripheral nerve lesions at common sites of entrapment, or a patchy axonal neuropathy of unknown etiology. In 1 patient a diagnosis of HNPP remains most likely. DNA analysis for the deletion of 17p11.2 is clearly useful in establishing the diagnosis of HNPP, which should be considered regardless of family history or clinical evidence of a generalized neuropathy, and in patients with multifocal neuropathies that do not conform to the classic clinical practice of HNPP.
我们对51例多灶性神经病患者进行了研究,以检测17号染色体p11.2区域的缺失情况,该缺失在遗传性压力易感性神经病(HNPP)家系中已有报道。在24例患者中检测到了该缺失,其中包括来自15个家系中14个家系的19例患者,基于临床、神经生理学和/或病理学依据,这些患者曾被认为很可能患有HNPP。1例存在该缺失的患者具有相当不寻常的HNPP临床特征,表现为进行性肩胛腓骨肌综合征。总体而言,19例存在该缺失的索引患者中,7例(37%)没有患病亲属,且不到一半的患者在检查时有全身性神经病的证据。在存在该缺失的患者中,仅15例(62%)的周围神经病变与受压有关。对25例患者及其亲属中的23例进行的神经传导研究显示,远端感觉和运动潜伏期适度延长、传导速度减慢以及感觉或诱发肌肉动作电位幅度不同程度降低,呈现出相当一致的模式。未检测到17p11.2缺失的被研究患者情况各异,包括患有复发性和/或家族性神经性肌萎缩、在常见卡压部位有两处或更多处周围神经病变,或病因不明的斑片状轴索性神经病的患者。1例患者最有可能诊断为HNPP。对17p11.2缺失进行DNA分析对于确立HNPP的诊断显然是有用的,无论有无家族史或全身性神经病的临床证据,对于不符合HNPP经典临床特征的多灶性神经病患者也应考虑进行该分析。