Yanagi Kumiko, Kaname Tadashi, Chinen Yasutsugu, Naritomi Kenji
Department of Medical Genetics, University of Ryukyus Graduate School of Medicine, Okinawa, Japan.
Congenit Anom (Kyoto). 2004 Sep;44(3):137-41. doi: 10.1111/j.1741-4520.2004.00026.x.
The human faciogenital dysplasia 1 (FGD1) gene product plays an important role in morphogenesis. Its dysfunction causes Aarskog-Scott syndrome (MIM musical sharp 305400). To characterize the FGD1, we investigated its expression by RT-PCR and Southern blot analysis in normal tissues. We found novel alternative forms of the FGD1. One has a novel exon located in intron 8, named exon 8B (8B FDG1) and the other has an exon in intron 7, exon 7B (7B FGD1). The 8B FDG1 is expressed strongly in the brain, testis, spinal cord, trachea and stomach, and weakly in the thymus and lymphocytes. However, expression of the 7B FGD1 is weak and restricted in the testis and salivary gland. Insertion of each novel exon results in production of a premature termination codon, respectively, and the predicted proteins generated from them have only a proline-rich domain and an incomplete DH domain which potentially compete with the wild type of FGD1.
人类面部生殖器发育异常1(FGD1)基因产物在形态发生中起重要作用。其功能障碍会导致Aarskog-Scott综合征(MIM编号305400)。为了表征FGD1,我们通过逆转录聚合酶链反应(RT-PCR)和Southern印迹分析研究了其在正常组织中的表达。我们发现了FGD1的新型可变形式。一种在位于内含子8中的新型外显子,命名为外显子8B(8B FDG1),另一种在内含子7中有一个外显子,外显子7B(7B FDG1)。8B FDG1在脑、睾丸、脊髓、气管和胃中强烈表达,在胸腺和淋巴细胞中弱表达。然而,7B FDG1的表达较弱且局限于睾丸和唾液腺。每个新型外显子的插入分别导致产生提前终止密码子,并且由此产生的预测蛋白质仅具有富含脯氨酸的结构域和不完整的DH结构域,这可能与野生型FGD1竞争。