当Rett综合征是由MECP2以外的基因引起时。

Alan K Percy, Jane Lane, Fran Annese, Hannah Warren, Steven A Skinner, Jeffrey L Neul
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引用次数: 19

摘要

两名符合Rett综合征(RTT)诊断标准但缺乏MECP2突变的患者接受了额外的基因检测。MECP2是与该疾病主要相关的基因。该检测揭示了与RTT无关的基因CTNNB1的致病性突变,该突变导致常染色体显性神经发育障碍,影响细胞信号传导和转录因子,以及WDR45基因的可能致病性突变,该突变与儿童早期发育迟缓和青春期或成年期与苍白球和黑质铁积累相关的进行性神经退行性变性有关。这两个个体的描述与先前的报道有关,这些报道将多个其他基因与RTT联系起来,但未能显示MECP2突变。这些个体强调,当没有检测到MECP2突变时,需要在RTT中进行额外的分子检测。
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When Rett syndrome is due to genes other than MECP2.

Two individuals meeting diagnostic criteria for Rett syndrome (RTT) but lacking a mutation in MECP2, the gene predominantly associated with this disorder, were provided additional genetic testing. This testing revealed pathogenic mutations in a gene not previously associated with RTT, CTNNB1, mutations in which lead to an autosomal dominant neurodevelopmental disorder affecting cell signaling and transcription factors as well as a likely pathogenic mutation in the WDR45 gene, which is associated with developmental delay in early childhood and progressive neurodegeneration in adolescence or adulthood related to iron accumulation in the globus pallidus and substantia nigra. These two individuals are described in relation to previous reports linking multiple other genes with RTT failing to show an MECP2 mutation. These individuals underscore the need to pursue additional molecular testing in RTT when a mutation in MECP2 is not detected.

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