Department of Medical Biology and Genetics, Akdeniz University Medical School, Antalya, Turkey.
Department of Pediatric Genetics, Akdeniz University Medical School, Antalya, Turkey.
Pediatr Neurol. 2019 Oct;99:7-15. doi: 10.1016/j.pediatrneurol.2019.01.018. Epub 2019 Feb 2.
Craniosynostosis is a pathologic craniofacial disorder and is defined as the premature fusion of one or more cranial (calvarial) sutures. Cranial sutures are fibrous joints consisting of nonossified mesenchymal cells that play an important role in the development of healthy craniofacial skeletons. Early fusion of these sutures results in incomplete brain development that may lead to complications of several severe medical conditions including seizures, brain damage, mental delay, complex deformities, strabismus, and visual and breathing problems. As a congenital disease, craniosynostosis has a heterogeneous origin that can be affected by genetic and epigenetic alterations, teratogens, and environmental factors and make the syndrome highly complex. To date, approximately 200 syndromes have been linked to craniosynostosis. In addition to being part of a syndrome, craniosynostosis can be nonsyndromic, formed without any additional anomalies. More than 50 nuclear genes that relate to craniosynostosis have been identified. Besides genetic factors, epigenetic factors like microRNAs and mechanical forces also play important roles in suture fusion. As craniosynostosis is a multifactorial disorder, evaluating the craniosynostosis syndrome requires and depends on all the information obtained from clinical findings, genetic analysis, epigenetic or environmental factors, or gene modulators. In this review, we will focus on embryologic and genetic studies, as well as epigenetic and environmental studies. We will discuss published studies and correlate the findings with unknown aspects of craniofacial disorders.
颅缝早闭是一种病理性颅面畸形,定义为一个或多个颅骨(颅骨)缝过早融合。颅骨缝是由未骨化的间充质细胞组成的纤维关节,在健康颅面骨骼的发育中起着重要作用。这些缝线的早期融合会导致大脑发育不完全,可能导致多种严重疾病的并发症,包括癫痫、脑损伤、智力迟钝、复杂畸形、斜视以及视觉和呼吸问题。作为一种先天性疾病,颅缝早闭具有遗传和表观遗传改变、致畸原和环境因素等多种异质性起源,使该综合征非常复杂。迄今为止,大约有 200 种综合征与颅缝早闭有关。除了作为综合征的一部分,颅缝早闭也可以是非综合征性的,即没有任何其他异常形成的。已经鉴定出 50 多个与颅缝早闭相关的核基因。除了遗传因素外,表观遗传因素,如 microRNAs 和机械力,也在缝线融合中发挥重要作用。由于颅缝早闭是一种多因素疾病,评估颅缝早闭综合征需要并取决于从临床发现、遗传分析、表观遗传或环境因素或基因调节剂中获得的所有信息。在这篇综述中,我们将重点介绍胚胎学和遗传学研究,以及表观遗传学和环境研究。我们将讨论已发表的研究,并将这些发现与颅面畸形的未知方面相关联。