Department of Biological Sciences, Lehigh University, Bethlehem, Pennsylvania, United States of America.
Department of Biological Sciences, Lehigh University, Bethlehem, Pennsylvania, United States of America ; Merck, Sharp & Dohme, West Point, Pennsylvania, United States of America.
PLoS Genet. 2013;9(12):e1004036. doi: 10.1371/journal.pgen.1004036. Epub 2013 Dec 19.
Roberts Syndrome (RBS) and Cornelia de Lange Syndrome (CdLS) are severe developmental maladies that present with nearly an identical suite of multi-spectrum birth defects. Not surprisingly, RBS and CdLS arise from mutations within a single pathway--here involving cohesion. Sister chromatid tethering reactions that comprise cohesion are required for high fidelity chromosome segregation, but cohesin tethers also regulate gene transcription, promote DNA repair, and impact DNA replication. Currently, RBS is thought to arise from elevated levels of apoptosis, mitotic failure, and limited progenitor cell proliferation, while CdLS is thought to arise, instead, from transcription dysregulation. Here, we review new information that implicates RBS gene mutations in altered transcription profiles. We propose that cohesin-dependent transcription dysregulation may extend to other developmental maladies; the diagnoses of which are complicated through multi-functional proteins that manifest a sliding scale of diverse and severe phenotypes. We further review evidence that cohesinopathies are more common than currently posited.
罗伯茨综合征(RBS)和科内利亚德朗热综合征(CdLS)是两种严重的发育障碍疾病,它们表现出几乎完全相同的多种出生缺陷。毫不奇怪,RBS 和 CdLS 是由单个途径中的突变引起的,在此过程中涉及黏合。组成黏合的姐妹染色单体连接反应对于高保真染色体分离是必需的,但黏合蛋白还调节基因转录、促进 DNA 修复和影响 DNA 复制。目前,人们认为 RBS 是由于细胞凋亡增加、有丝分裂失败和祖细胞增殖有限引起的,而 CdLS 则被认为是由于转录失调引起的。在这里,我们回顾了新的信息,表明 RBS 基因突变与转录谱的改变有关。我们提出,黏合蛋白依赖性转录失调可能会扩展到其他发育障碍疾病;这些疾病的诊断因表现出多种严重表型的多功能蛋白而变得复杂。我们进一步回顾了黏合蛋白病比目前认为的更为常见的证据。