Faculty of Medicine and Dentistry, University of Alberta, Edmonton, AB, Canada.
Blueprint Genetics, A Quest Diagnostics Company, Espoo, Finland.
Eur J Med Genet. 2023 Jan;66(1):104657. doi: 10.1016/j.ejmg.2022.104657. Epub 2022 Nov 9.
The RFX7 gene is one of eight genes within the regulatory factor X family. RFX7 is highly expressed in the brain and plays an important role in cell maturation and differentiation. It has only recently been implicated in disease in humans. Reports from 15 individuals have described RFX-associated phenotype as a neurobehavioural disease, manifesting primarily with global developmental delay and intellectual disability. Autism spectrum disorder and attention deficit hyperactivity disorder have also been described in some children. Here we report a case of a 19-month-old with a de novo missense variant in RFX7 resulting in severe global developmental delay including significant speech delay, microcephaly, dyskinetic movements, and failure to thrive. This is the first association between variants in RFX7 and failure to thrive, expanding the phenotype of this newly described gene. In this report we will also show RFX7 associated progressive central nervous system involvement through serial brain imaging.
RFX7 基因是调节因子 X 家族的 8 个基因之一。RFX7 在大脑中高度表达,在细胞成熟和分化中发挥重要作用。它最近才被发现与人类疾病有关。来自 15 个人的报告描述了与 RFX 相关的表型为神经行为疾病,主要表现为全面发育迟缓伴智力残疾。一些儿童也被描述为自闭症谱系障碍和注意缺陷多动障碍。在这里,我们报告了一例 19 个月大的儿童,其 RFX7 中存在新生错义变异,导致严重的全面发育迟缓,包括严重的语言发育迟缓、小头畸形、运动障碍和生长不良。这是 RFX7 变异与生长不良之间的首次关联,扩展了这个新描述基因的表型。在本报告中,我们还将通过连续脑成像显示与 RFX7 相关的进行性中枢神经系统受累。