Two Cases of Primary Ciliary Dyskinesia with Different Responses to Macrolide Treatment

Takashi Kido; Kazuhiro Yatera; Kei Yamasaki; Shuya Nagata; Yasuo Choujin; Chiyo Yamaga; Kanako Hara; Hiroshi Ishimoto; Masanori Hisaoka; Hiroshi Mukae

doi:10.2169/internalmedicine.51.6617

CASE REPORTS

Two Cases of Primary Ciliary Dyskinesia with Different Responses to Macrolide Treatment

Takashi Kido, Kazuhiro Yatera, Kei Yamasaki, Shuya Nagata, Yasuo Choujin, Chiyo Yamaga, Kanako Hara, Hiroshi Ishimoto, Masanori Hisaoka, Hiroshi Mukae

Author information

Takashi Kido
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan
Kazuhiro Yatera
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan
Kei Yamasaki
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan
Shuya Nagata
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan
Yasuo Choujin
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan
Chiyo Yamaga
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan
Kanako Hara
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan
Hiroshi Ishimoto
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan
Masanori Hisaoka
Department of Pathology and Oncology, University of Occupational and Environmental Health, Japan
Hiroshi Mukae
Department of Respiratory Medicine, University of Occupational and Environmental Health, Japan

Keywords: azithromycin, clarithromycin, dynein arm, immotile cilia, macrolide, primary ciliary dyskinesia

JOURNAL OPEN ACCESS

2012 Volume 51 Issue 9 Pages 1093-1098

DOI https://doi.org/10.2169/internalmedicine.51.6617

Details

Abstract

We herein report two cases of primary ciliary dyskinesia (PCD) with different responses to macrolides. Case 1: a 17-year-old Japanese man with Pseudomonas aeruginosa infection and combined defect of both inner and outer dynein arms in the cilia was unsuccessfully treated with long-term macrolides (clarithromycin, erythromycin, and azithromycin). Case 2: a 70-year-old Japanese man with deficiency of only the inner dynein arm was successfully treated with clarithromycin. Though the reasons for the different responses to macrolides are unclear, differences of ultrastructural abnormalities of the cilia might be one of the predictive factors in PCD just as in Pseudomonas aeruginosa infection.

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