Swerdlow R H, Wooten G F
Department of Neurology and the Center for the Study of Neurodegenerative Diseases, University of Virginia Health System, Charlottesville, USA.
Ann Neurol. 2001 Oct;50(4):537-40. doi: 10.1002/ana.1160.
Sex-linked male deafness and dystonia (Mohr-Tranebjaerg syndrome) arises from mutation of the deafness/dystonia peptide (DDP) gene. We describe a novel guanine deletion at nucleotide 108 of the DDP gene in a family with Mohr-Tranebjaerg syndrome, which terminates this 97-amino acid protein at codon 25. Unlike previously reported kindreds, carrier females in this family also manifest dystonias, including torticollis and writer's cramp. A family history of male deafness should alert clinicians to the possibility of DDP mutation in women with focal dystonias.
性连锁型男性耳聋和肌张力障碍(莫尔-特拉内耶尔格综合征)源于耳聋/肌张力障碍肽(DDP)基因突变。我们在一个患有莫尔-特拉内耶尔格综合征的家族中描述了DDP基因第108位核苷酸处一个新的鸟嘌呤缺失,该缺失使这个97个氨基酸的蛋白质在第25密码子处终止。与先前报道的家族不同,这个家族中的女性携带者也表现出肌张力障碍,包括斜颈和书写痉挛。男性耳聋的家族史应提醒临床医生注意患有局灶性肌张力障碍的女性存在DDP突变的可能性。