Camus P, Abbadi N, Perrier M C, Chéry M, Gilgenkrantz S
Laboratoire de Génétique, C.H.U. de Brabois, Vandoeuvre-Lès-Nancy, France.
Hum Genet. 1996 Feb;97(2):247-50. doi: 10.1007/BF02265275.
Rett syndrome (RS) is a neurologic disorder with an exclusive incidence in females. A nonrandom X-inactivation could provide insight into the understanding of this disease. We performed molecular analysis based on the differential methylation of the active and inactive X with probe M27 beta, taking into account the parental origin of the two Xs, in 30 control girls, 8 sisters, and 30 RS girls. In 27 control an 31 RS mothers, the inactivation status of the X transmitted to their daughters was also analyzed. The results showed a significantly increased frequency of partial paternal X inactivation (> 65%) in lymphocytes from 16/30 RS compared with 4/30 controls (P = 0.001). These results do not support the hypothesis of a monogenic X-linked mutation but should be taken into account when researching the etiology of this disease.
瑞特综合征(RS)是一种仅在女性中发病的神经疾病。非随机X染色体失活有助于深入了解这种疾病。我们使用探针M27β,基于活性和非活性X染色体的差异甲基化进行分子分析,同时考虑两条X染色体的亲本来源,研究对象包括30名对照女孩、8名姐妹以及30名患RS的女孩。我们还分析了27名对照母亲和31名患RS的母亲传递给女儿的X染色体的失活状态。结果显示,与4/30的对照相比,16/30的患RS者淋巴细胞中部分父源X染色体失活的频率显著增加(>65%)(P = 0.001)。这些结果不支持单基因X连锁突变的假说,但在研究该疾病的病因时应予以考虑。