{"title":"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","authors":"Akira Umeda, Hidenori Kiyosawa, Kazuya Miyagawa, Yoshiyuki Ohira, Masataka Higuchi, Ho Namkoong, Takanori Asakura, Makoto Ishii, Naoki Hasegawa, Koichi Fukunaga","doi":"10.1016/j.resinv.2025.09.023","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":20934,"journal":{"name":"Respiratory investigation","volume":"63 6","pages":"1243-1245"},"PeriodicalIF":2.0000,"publicationDate":"2025-10-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"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.\",\"authors\":\"Akira Umeda, Hidenori Kiyosawa, Kazuya Miyagawa, Yoshiyuki Ohira, Masataka Higuchi, Ho Namkoong, Takanori Asakura, Makoto Ishii, Naoki Hasegawa, Koichi Fukunaga\",\"doi\":\"10.1016/j.resinv.2025.09.023\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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.</p>\",\"PeriodicalId\":20934,\"journal\":{\"name\":\"Respiratory investigation\",\"volume\":\"63 6\",\"pages\":\"1243-1245\"},\"PeriodicalIF\":2.0000,\"publicationDate\":\"2025-10-08\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Respiratory investigation\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1016/j.resinv.2025.09.023\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"RESPIRATORY SYSTEM\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Respiratory investigation","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1016/j.resinv.2025.09.023","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"RESPIRATORY SYSTEM","Score":null,"Total":0}
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.