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KMT2A突变所致维德曼-施泰纳综合征的临床特征描述。

Delineation of clinical features in Wiedemann-Steiner syndrome caused by KMT2A mutations.

作者信息

Miyake N, Tsurusaki Y, Koshimizu E, Okamoto N, Kosho T, Brown N J, Tan T Y, Yap P J J, Suzumura H, Tanaka T, Nagai T, Nakashima M, Saitsu H, Niikawa N, Matsumoto N

机构信息

Department of Human Genetics, Yokohama City University Graduate School of Medicine, Yokohama, Japan.

Department of Medical Genetics, Osaka Medical Center and Research Institute for Maternal and Child Health, Izumi, Japan.

出版信息

Clin Genet. 2016 Jan;89(1):115-9. doi: 10.1111/cge.12586. Epub 2015 Apr 14.

Abstract

Wiedemann-Steiner syndrome (WSS) is an autosomal dominant congenital anomaly syndrome characterized by hairy elbows, dysmorphic facial appearances (hypertelorism, thick eyebrows, downslanted and vertically narrow palpebral fissures), pre- and post-natal growth deficiency, and psychomotor delay. WSS is caused by heterozygous mutations in KMT2A (also known as MLL), a gene encoding a histone methyltransferase. Here, we identify six novel KMT2A mutations in six WSS patients, with four mutations occurring de novo. Interestingly, some of the patients were initially diagnosed with atypical Kabuki syndrome, which is caused by mutations in KMT2D or KDM6A, genes also involved in histone methylation. KMT2A mutations and clinical features are summarized in our six patients together with eight previously reported patients. Furthermore, clinical comparison of the two syndromes is discussed in detail.

摘要

维德曼-施泰纳综合征(WSS)是一种常染色体显性先天性异常综合征,其特征为肘部多毛、面部畸形(眼距过宽、眉毛浓密、睑裂向下倾斜且垂直变窄)、产前和产后生长发育迟缓以及精神运动发育迟缓。WSS由KMT2A(也称为MLL)基因的杂合突变引起,该基因编码一种组蛋白甲基转移酶。在此,我们在6例WSS患者中鉴定出6种新的KMT2A突变,其中4种突变为新发突变。有趣的是,部分患者最初被诊断为非典型歌舞伎综合征,该综合征由KMT2D或KDM6A基因的突变引起,这两个基因也参与组蛋白甲基化。我们将6例患者以及之前报道的8例患者的KMT2A突变和临床特征进行了总结。此外,还详细讨论了这两种综合征的临床比较。

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