BACKGROUND: The diagnosis of primary ciliary dyskinesia (PCD) can be challenging, and it may be particularly difficult to distinguish primary ciliary disease from the secondary changes after infections. OBJECTIVES: The purpose of the study was to evaluate if nasal epithelial cells, obtained with nasal brushing instead of a biopsy, could be used in a culture system for the diagnosis of PCD in difficult cases. METHODS AND MAIN RESULTS: Ciliary motion analysis (CMA) and transmission electron microscopy (TEM) were performed on 59 subjects with persistent or recurrent pneumonia. These investigations allowed the diagnosis of PCD in 13 (22%) patients while the defect of the cilia was considered secondary to infections in 37 (63%) subjects. In the remaining nine (15%) patients the diagnostic evaluation with CMA and TEM remained inconclusive. Ciliogenesis in culture allowed the diagnosis of PCD in four of these patients, it was indicative of a secondary defect in two subjects, and it was not helpful in the remaining three patients. CONCLUSIONS: Culture of cells obtained with brushing of the nasal turbinate is not a perfect test, nevertheless it may offer diagnostic help in doubtful cases of PCD.
BACKGROUND: The diagnosis of primary ciliary dyskinesia (PCD) can be challenging, and it may be particularly difficult to distinguish primary ciliary disease from the secondary changes after infections. OBJECTIVES: The purpose of the study was to evaluate if nasal epithelial cells, obtained with nasal brushing instead of a biopsy, could be used in a culture system for the diagnosis of PCD in difficult cases. METHODS AND MAIN RESULTS: Ciliary motion analysis (CMA) and transmission electron microscopy (TEM) were performed on 59 subjects with persistent or recurrent pneumonia. These investigations allowed the diagnosis of PCD in 13 (22%) patients while the defect of the cilia was considered secondary to infections in 37 (63%) subjects. In the remaining nine (15%) patients the diagnostic evaluation with CMA and TEM remained inconclusive. Ciliogenesis in culture allowed the diagnosis of PCD in four of these patients, it was indicative of a secondary defect in two subjects, and it was not helpful in the remaining three patients. CONCLUSIONS: Culture of cells obtained with brushing of the nasal turbinate is not a perfect test, nevertheless it may offer diagnostic help in doubtful cases of PCD.
Authors: Jane S Lucas; Angelo Barbato; Samuel A Collins; Myrofora Goutaki; Laura Behan; Daan Caudri; Sharon Dell; Ernst Eber; Estelle Escudier; Robert A Hirst; Claire Hogg; Mark Jorissen; Philipp Latzin; Marie Legendre; Margaret W Leigh; Fabio Midulla; Kim G Nielsen; Heymut Omran; Jean-Francois Papon; Petr Pohunek; Beatrice Redfern; David Rigau; Bernhard Rindlisbacher; Francesca Santamaria; Amelia Shoemark; Deborah Snijders; Thomy Tonia; Andrea Titieni; Woolf T Walker; Claudius Werner; Andrew Bush; Claudia E Kuehni Journal: Eur Respir J Date: 2017-01-04 Impact factor: 16.671
Authors: Lindsay Broadbent; Sheerien Manzoor; Maria C Zarcone; Judit Barabas; Michael D Shields; Sejal Saglani; Claire M Lloyd; Andrew Bush; Adnan Custovic; Peter Ghazal; Mindy Gore; Ben Marsland; Graham Roberts; Jurgen Schwarze; Steve Turner; Ultan F Power Journal: PLoS One Date: 2020-03-26 Impact factor: 3.240