Abbas N, Lücking C B, Ricard S, Dürr A, Bonifati V, De Michele G, Bouley S, Vaughan J R, Gasser T, Marconi R, Broussolle E, Brefel-Courbon C, Harhangi B S, Oostra B A, Fabrizio E, Böhme G A, Pradier L, Wood N W, Filla A, Meco G, Denefle P, Agid Y, Brice A
INSERM U289, Hôpital de la Salpêtrière, 47 Boulevard de l'Hôpital, 75651 Paris Cedex 13, France.
Hum Mol Genet. 1999 Apr;8(4):567-74. doi: 10.1093/hmg/8.4.567.
Autosomal recessive juvenile parkinsonism (AR-JP, PARK2; OMIM 602544), one of the monogenic forms of Parkinson's disease (PD), was initially described in Japan. It is characterized by early onset (before age 40), marked response to levodopa treatment and levodopa-induced dyskinesias. The gene responsible for AR-JP was recently identified and designated parkin. We have analysed the 12 coding exons of the parkin gene in 35 mostly European families with early onset autosomal recessive parkinsonism. In one family, a homozygous deletion of exon 4 could be demonstrated. By direct sequencing of the exons in the index patients of the remaining 34 families, eight previously undescribed point mutations (homozygous or heterozygous) were detected in eight families that included 20 patients. The mutations segregated with the disease in the families and were not detected on 110-166 control chromosomes. Four mutations caused truncation of the parkin protein. Three were frameshifts (202-203delAG, 255delA and 321-322insGT) and one a nonsense mutation (Trp453Stop). The other four were missense mutations (Lys161Asn, Arg256Cys, Arg275Trp and Thr415Asn) that probably affect amino acids that are important for the function of the parkin protein, since they result in the same phenotype as truncating mutations or homozygous exon deletions. Mean age at onset was 38 +/- 12 years, but onset up to age 58 was observed. Mutations in the parkin gene are therefore not invariably associated with early onset parkinsonism. In many patients, the phenotype is indistinguishable from that of idiopathic PD. This study has shown that a wide variety of different mutations in the parkin gene are a common cause of autosomal recessive parkinsonism in Europe and that different types of point mutations seem to be more frequently responsible for the disease phenotype than are deletions.
常染色体隐性少年型帕金森病(AR-JP,PARK2;OMIM 602544)是帕金森病(PD)的单基因形式之一,最初在日本被描述。其特征为发病早(40岁之前)、对左旋多巴治疗反应明显以及左旋多巴诱导的运动障碍。负责AR-JP的基因最近已被鉴定并命名为帕金蛋白基因(parkin)。我们分析了35个主要为欧洲家族的帕金蛋白基因的12个编码外显子,这些家族患有早发性常染色体隐性帕金森病。在一个家族中,证实存在外显子4的纯合缺失。通过对其余34个家族的索引患者的外显子进行直接测序,在包括20名患者的8个家族中检测到8个先前未描述的点突变(纯合或杂合)。这些突变在家族中与疾病共分离,并且在110 - 166条对照染色体上未检测到。四个突变导致帕金蛋白截短。三个是移码突变(202 - 203delAG、255delA和321 - 322insGT),一个是无义突变(Trp453Stop)。其他四个是错义突变(Lys161Asn、Arg256Cys、Arg275Trp和Thr415Asn),可能影响对帕金蛋白功能重要的氨基酸,因为它们导致与截短突变或纯合外显子缺失相同的表型。平均发病年龄为38±12岁,但观察到发病年龄可达58岁。因此,帕金蛋白基因突变并非总是与早发性帕金森病相关。在许多患者中,其表型与特发性帕金森病难以区分。这项研究表明,帕金蛋白基因中广泛的不同突变是欧洲常染色体隐性帕金森病的常见原因,并且不同类型的点突变似乎比缺失更常导致疾病表型。