双等位基因c.2709del和c.3020T >g引起dnah11相关的原发性纤毛运动障碍,表现为Kartagener综合征:气道上皮细胞群弥漫性扭曲波状运动的可能新表型

IF 2 Q2 RESPIRATORY SYSTEM
Akira Umeda, Hidenori Kiyosawa, Kazuya Miyagawa, Yoshiyuki Ohira, Masataka Higuchi, Ho Namkoong, Takanori Asakura, Makoto Ishii, Naoki Hasegawa, Koichi Fukunaga
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引用次数: 0

摘要

原发性纤毛运动障碍(PCD)是一种罕见的遗传性疾病与纤毛功能障碍。逆位、慢性鼻窦炎和支气管扩张的三联征被称为卡塔赫纳综合征。我们报告了一名PCD患者,其表现为Kartagener综合征,具有DNAH11等位基因的新组合,即移码变体c.2709del p.(Trp904Glyfs * 5)和无意义变体c.3020T >gp .(Leu1007 *)。双等位基因致病性变异罕见且位于n端附近。高速视频显微镜显示不一致的纤毛和气道上皮细胞群的弥漫性扭曲波状运动。研究者应该注意到这一现象,并需要进一步的研究来阐明DNAH11在PCD中的作用。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Biallelic c.2709del and c.3020T>G cause DNAH11-related primary ciliary dyskinesia presenting with Kartagener syndrome: Possible novel phenotype of diffuse-twisting wave-like movements of airway epithelial cell populations.

Primary ciliary dyskinesia (PCD) is a rare genetic disorder with ciliary dysfunction. The triad of situs inversus, chronic sinusitis, and bronchiectasis is termed Kartagener syndrome. We report a PCD patient presenting with Kartagener syndrome with a novel combination of DNAH11 alleles, i.e., a frameshift variant, c.2709del p.(Trp904Glyfs∗5), and a nonsense variant, c.3020T>G p.(Leu1007∗). The biallelic pathogenic variants were both rare and located close to the N-terminus. High-speed video microscopy revealed discordant cilia and diffuse-twisting wave-like movements of the airway epithelial cell populations. Researchers should note this phenomenon, and further investigation is necessary to clarify the role of DNAH11 in PCD.

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来源期刊
Respiratory investigation
Respiratory investigation RESPIRATORY SYSTEM-
CiteScore
4.90
自引率
6.50%
发文量
114
审稿时长
64 days
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