Genomics Coordination Center, Department of Genetics, University Medical Center Groningen, University of Groningen, The Netherlands.
Hum Mutat. 2013 Dec;34(12):1597-605. doi: 10.1002/humu.22440. Epub 2013 Oct 16.
Microvillus inclusion disease (MVID) is one of the most severe congenital intestinal disorders and is characterized by neonatal secretory diarrhea and the inability to absorb nutrients from the intestinal lumen. MVID is associated with patient-, family-, and ancestry-unique mutations in the MYO5B gene, encoding the actin-based motor protein myosin Vb. Here, we review the MYO5B gene and all currently known MYO5B mutations and for the first time methodologically categorize these with regard to functional protein domains and recurrence in MYO7A associated with Usher syndrome and other myosins. We also review animal models for MVID and the latest data on functional studies related to the myosin Vb protein. To congregate existing and future information on MVID geno-/phenotypes and facilitate its quick and easy sharing among clinicians and researchers, we have constructed an online MOLGENIS-based international patient registry (www.MVID-central.org). This easily accessible database currently contains detailed information of 137 MVID patients together with reported clinical/phenotypic details and 41 unique MYO5B mutations, of which several unpublished. The future expansion and prospective nature of this registry is expected to improve disease diagnosis, prognosis, and genetic counseling.
微绒毛包涵体病(MVID)是最严重的先天性肠病之一,其特征是新生儿分泌性腹泻和无法从肠腔吸收营养。MVID 与 MYO5B 基因的患者、家族和祖先独特的突变有关,该基因编码基于肌动蛋白的运动蛋白肌球蛋白 Vb。在这里,我们回顾了 MYO5B 基因和所有目前已知的 MYO5B 突变,并首次根据功能蛋白结构域和与 Usher 综合征和其他肌球蛋白相关的 MYO7A 中的复发对其进行了分类。我们还回顾了 MVID 的动物模型和与肌球蛋白 Vb 蛋白相关的最新功能研究数据。为了汇集现有的和未来的关于 MVID 基因型/表型的信息,并促进临床医生和研究人员之间的快速和容易的共享,我们构建了一个基于在线 MOLGENIS 的国际患者登记处(www.MVID-central.org)。这个易于访问的数据库目前包含了 137 名 MVID 患者的详细信息,以及报告的临床/表型细节和 41 个独特的 MYO5B 突变,其中一些是未发表的。该登记处的未来扩展和前瞻性预计将改善疾病诊断、预后和遗传咨询。