Pieras Juan I, Muñoz-Cabello Beatriz, Borrego Salud, Marcos Irene, Sanchez Javier, Madruga Marcos, Antiñolo Guillermo
Unidad de Gestión Clínica de Genética, Reproducción y Medicina Fetal, Instituto de Biomedicina de Sevilla (IBIS), Hospital Universitario Virgen del Rocío/CSIC/Universidad de Sevilla, Spain.
Brain Dev. 2011 Aug;33(7):608-11. doi: 10.1016/j.braindev.2010.09.012. Epub 2010 Oct 22.
Rett Syndrome (RS; MIM_312750) is a severe and progressive neurodevelopmental disorder affecting principally females. Mutations in X-Linked MECP2 gene (methyl CpG-binding protein 2; MIM_300005) have been reported as being the major cause of RS. Mutations in this gene have been described as cause of wide spectrum of neurological disorders and mental retardation in males. In some cases, mutations in MECP2 in males produce clinical picture similar to RS. Here we report the identification of the novel truncating mutation Y120X in a 4-year-old child with atypical RS phenotype. Chromosome analysis showed a normal karyotype, and blood DNA and tissue DNA analysis reveal a mosaic for the mutation. Patient's mother DNA analysis showed that this is a de novo mutation, that has never been described before in any female or male case of RS.
雷特综合征(RS;MIM_312750)是一种严重的进行性神经发育障碍,主要影响女性。据报道,X连锁MECP2基因(甲基CpG结合蛋白2;MIM_300005)的突变是RS的主要病因。该基因的突变被认为是男性广泛的神经疾病和智力迟钝的病因。在某些情况下,男性MECP2基因的突变会产生与RS相似的临床表现。在此,我们报告在一名具有非典型RS表型的4岁儿童中鉴定出新型截短突变Y120X。染色体分析显示核型正常,血液DNA和组织DNA分析揭示该突变呈嵌合状态。对患者母亲的DNA分析表明,这是一种新发突变,在任何RS女性或男性病例中均未曾描述过。