LSUHSC Department of Pediatrics, New Orleans, Louisiana, USA.
Tulane University School of Medicine, New Orleans, Louisiana, USA.
Am J Med Genet A. 2022 Dec;188(12):3535-3539. doi: 10.1002/ajmg.a.62964. Epub 2022 Sep 7.
Short stature, hearing loss, retinitis pigmentosa, and distinctive facies (SHRF) Syndrome is a syndrome recently identified among three German patients. Clinical characteristics include eye disease, sensorineural hearing loss, distinct facial and phalangeal features, short stature, developmental delay, and cerebellar atrophy. In this case report, we discuss a fourth identified patient with genomic mutations in the EXOSC2 gene which codes for a cap protein in the RNA exosome. Whole exome sequencing identified two mutations of unknown clinical significance including: a heterozygous maternal variant, missense mutation NM_014285.7: c427G>A (p.Ala143Thr) in exon 6 and a heterozygous paternal variant, splice donor NM_014285.5: c.801+1G>A in intron 8. Our patient demonstrates a novel clinical presentation within the SHRF disease spectrum.
短身材、听力损失、视网膜色素变性和特殊面容(SHRF)综合征是最近在三名德国患者中发现的一种综合征。临床特征包括眼部疾病、感觉神经性听力损失、独特的面部和指骨特征、身材矮小、发育迟缓以及小脑萎缩。在本病例报告中,我们讨论了第四位患者,其 EXOSC2 基因存在导致 RNA 外切体帽蛋白的基因突变。全外显子组测序发现了两种具有未知临床意义的突变,包括:杂合的母体变异,错义突变 NM_014285.7:c427G>A(p.Ala143Thr)在第 6 外显子和杂合的父系变体,剪接供体 NM_014285.5:c.801+1G>A 在内含子 8。我们的患者在 SHRF 疾病谱中表现出一种新的临床表现。