Motch Perrine Susan M, Wu Meng, Holmes Greg, Bjork Bryan C, Jabs Ethylin Wang, Richtsmeier Joan T
Department of Anthropology, The Pennsylvania State University, University Park, PA 16802, USA.
Department of Genetics and Genomic Sciences, Icahn School of Medicine at Mount Sinai, New York, NY 10029, USA.
J Dev Biol. 2020 Dec 5;8(4):30. doi: 10.3390/jdb8040030.
The phenotype currently accepted as Pierre Robin syndrome/sequence/anomalad/complex (PR) is characterized by mandibular dysmorphology, glossoptosis, respiratory obstruction, and in some cases, cleft palate. A causative sequence of developmental events is hypothesized for PR, but few clear causal relationships between discovered genetic variants, dysregulated gene expression, precise cellular processes, pathogenesis, and PR-associated anomalies are documented. This review presents the current understanding of PR phenotypes, the proposed pathogenetic processes underlying them, select genes associated with PR, and available animal models that could be used to better understand the genetic basis and phenotypic variation of PR.
目前被认定为皮埃尔·罗宾综合征/序列/异常/复合体(PR)的表型特征为下颌骨发育异常、舌后坠、呼吸阻塞,在某些情况下还伴有腭裂。针对PR提出了一系列发育事件的因果顺序假说,但已记录的已发现基因变异、基因表达失调、精确细胞过程、发病机制和PR相关异常之间明确的因果关系却很少。本综述介绍了目前对PR表型的理解、其潜在的发病机制、与PR相关的特定基因,以及可用于更好理解PR遗传基础和表型变异的现有动物模型。