Matijević Tanja, Knezević Jelena, Barisić Ingeborg, Resić Biserka, Culić Vida, Pavelić Jasminka
Rudjer Bosković Institute, Division of Molecular Medicine, Laboratory of Molecular Oncology, Bijenicka 54, 10002 Zagreb, Croatia.
Ann N Y Acad Sci. 2006 Dec;1091:225-32. doi: 10.1196/annals.1378.069.
Rett syndrome (RTT) is an X-linked dominant neurodevelopmental disorder almost exclusively affecting females and is usually sporadic. Mutations in MECP2 gene have been found in more than 80% of females with typical features of RTT. In this study, we analyzed 15 sporadic cases of RTT. In 7 of 15 patients (47%), we detected pathogenic mutations in the coding parts of MECP2 fourth exon. We found two missense (T158M, R133C), two nonsense (R168X, R270X), two frameshift mutations (P217fs and a double deletion of 28-bp at 1132-1159 and 10-bp at 1167-1176), and one in-frame deletion (L383_E392del10). To our knowledge, the last two mutations have not been reported yet. We also detected one previously described polymorphism (S194S). In conclusion, these results show that the fourth exon should be the first one analyzed because it harbors most of the known mutations. Moreover, mutation-negative cases should be further analyzed for gross rearrangements. This is the first study of its kind in Croatia and it enabled us to give the patients an early confirmation of RTT diagnosis.
瑞特综合征(RTT)是一种X连锁显性神经发育障碍,几乎只影响女性,通常为散发性。超过80%具有典型RTT特征的女性中发现了MECP2基因突变。在本研究中,我们分析了15例散发性RTT病例。在15例患者中的7例(47%),我们在MECP2第四外显子的编码部分检测到致病突变。我们发现了两个错义突变(T158M、R133C)、两个无义突变(R168X、R270X)、两个移码突变(P217fs以及1132 - 1159处28个碱基对和1167 - 1176处10个碱基对的双缺失),以及一个框内缺失(L383_E392del10)。据我们所知,最后两个突变尚未见报道。我们还检测到一个先前描述的多态性(S194S)。总之,这些结果表明第四外显子应作为首先分析的对象,因为它包含了大多数已知突变。此外,对于突变阴性的病例应进一步分析是否存在大片段重排。这是克罗地亚首例此类研究,它使我们能够为患者提供RTT诊断的早期确认。