Wawrocka Anna, Krawczynski Maciej R
Department of Medical Genetics, Poznan University of Medical Sciences, Rokietnicka 8, 60-806, Poznan, Poland.
Centers for Medical Genetics GENESIS, Poznan, Poland.
J Appl Genet. 2018 May;59(2):151-159. doi: 10.1007/s13353-017-0426-1. Epub 2018 Feb 19.
Aniridia is a rare, panocular disorder characterized by a variable degree of hypoplasia or the absence of iris tissue associated with additional ocular abnormalities. It is inherited in an autosomal dominant manner, with high penetrance and variable expression even within the same family. In most cases the disease is caused by haploinsufficiency truncating mutations in the PAX6 gene; however, in up to 30% of aniridia patients, disease results from chromosomal rearrangements at the 11p13 region. The aim of this review is to present the clinical and genetic aspects of the disease. Furthermore, we present a molecular diagnostic strategy in the aniridia patients. Recent improvement in the genetic diagnostic approach will precisely diagnosis aniridia patients, which is essential especially for children with aniridia in order to determine the risk of developing a Wilms tumor or neurodevelopmental disorder. Finally, based on the previous studies we describe the current knowledge and latest research findings in the topic of pathogenesis of aniridia and possible future treatment.
无虹膜症是一种罕见的全眼球疾病,其特征为不同程度的虹膜组织发育不全或缺失,并伴有其他眼部异常。它以常染色体显性方式遗传,具有高外显率,甚至在同一家族中也有不同的表现。在大多数情况下,该疾病是由PAX6基因的单倍体不足截断突变引起的;然而,在高达30%的无虹膜症患者中,疾病是由11p13区域的染色体重排导致的。本综述的目的是介绍该疾病的临床和遗传方面。此外,我们还介绍了针对无虹膜症患者的分子诊断策略。基因诊断方法的最新进展将精确诊断无虹膜症患者,这对于患有无虹膜症的儿童尤为重要,以便确定患威尔姆斯瘤或神经发育障碍的风险。最后,基于先前的研究,我们描述了无虹膜症发病机制主题的当前知识和最新研究发现以及可能的未来治疗方法。