Analysis of clinical and genetic features in an adolescent patient with primary ciliary dyskinesia induced by homozygous mutation in the RSPH4A gene: a case report

对一例由RSPH4A基因纯合突变引起的原发性纤毛运动障碍青少年患者的临床和遗传特征进行分析:病例报告

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Abstract

Primary ciliary dyskinesia (PCD) is a rare genetically heterogeneous disorder characterized by dysfunctional motile cilia, with or without detectable ultrastructural abnormalities. This study focuses on a homozygous mutation in the rare radial spoke head component 4A (RSPH4A) gene in a Chinese adolescent girl with PCD. The patient, an 11-year and 3-month-old girl, developed neonatal pneumonia after birth and gradually presented with persistent perennial rhinitis and recurrent productive cough. Lung CT scan indicated bronchiectasis, and whole-exome sequencing (WES) exhibited a novel pathogenic homozygous c.351dup (p. Pro118Serfs*2) frameshift mutation in the RSPH4A gene. A literature review reported that 21 pathogenic variants in RSPH4A have been discovered. WES recognized disease-causing mutations in PCD, and c.351dup (p. Pro118Serfs*2) frameshift mutation in RSPH4A may become a hotspot in Chinese patients.

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