A Novel Homozygous Missense Variant in the LRRC32 Gene Is Associated With a New Syndrome of Cleft Palate, Progressive Vitreoretinopathy, Growth Retardation, and Developmental Delay

错义突变 医学 外显子组测序 语音延迟 全球发育迟缓 遗传学 早产儿视网膜病变 FLNA公司 生物信息学 突变 儿科 基因 生物 胎龄 怀孕 表型 细胞骨架 细胞 菲拉明
作者
Zufit Hexner‐Erlichman,Boris Fichtman,Yoav Zehavi,Morad Khayat,Haneen Jabaly-Habib,Lee S. Izhaki-Tavor,Moshe Dessau,Orly Elpeleg,Ronen Spiegel
出处
期刊:Frontiers in Pediatrics [Frontiers Media]
卷期号:10: 859034-859034 被引量:5
标识
DOI:10.3389/fped.2022.859034
摘要

Cleft lip and/or cleft palate are a common group of birth defects that further classify into syndromic and non-syndromic forms. The syndromic forms are usually accompanied by additional physical or cognitive abnormalities. Isolated cleft palate syndromes are less common; however, they are associated with a variety of congenital malformations and generally have an underlying genetic etiology. A single report in 2019 described a novel syndrome in three individuals, characterized by cleft palate, developmental delay and proliferative retinopathy due to a homozygous non-sense mutation in the LRRC32 gene encoding glycoprotein A repetitions predominant (GARP), a cell surface polypeptide crucial for the processing and maturation of transforming growth factor β (TGF-β). We describe a patient who presented with cleft palate, prenatal and postnatal severe growth retardation, global developmental delay, dysmorphic facial features and progressive vitreoretinopathy. Whole exome sequencing (WES) revealed a very rare homozygous missense variant in the LRRC32 gene, which resulted in substitution of a highly conserved isoleucine to threonine. Protein modeling suggested this variant may negatively affect GARP function on latent TGF-β activation. In summary, our report further expands the clinical features of cleft palate, proliferative retinopathy and developmental delay syndrome and emphasizes the association of LRRC32 pathogenic variants with this new syndrome.
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