Associations between respiratory pathogens and lung function in primary ciliary dyskinesia: cross-sectional analysis from the PROVALF-PCD cohort.
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Rubbo Bruna, Kant Avni, Zhang Kewei, Allegorico Annalisa, Basilicata Simona, Boon Mieke, Borrelli Melissa, Calogero Claudia, Carr Siobhán B, Carroll Mary, Constant Carolina, Castillo Corullón Silvia, Corvol Harriet, Cutrera Renato, Dillenhöfer Stefanie, Emiralioglu Nagehan, Eralp Ela, Eryilmaz Polat Sanem, Gardner Laura, Gokdemir Yasemin, Harris Amanda, Hogg Claire, Karadag Bulent, Kobbernagel Helene, Koerner-Rettberg Cordula, Kouis Panayiotis, Lorent Natalie, Marcou Markella, Mathin June K, Martinu Vendula, Moreno-Galdó Antonio, Morgan Lucy, Nielsen Kim G, Omran Heymut, Ozcelik Ugur, Pohunek Petr, Raidt Johanna, Robinson Phil, Rovira-Amigo Sandra, Santamaria Francesca, Schlegtendal Anne, Tamalet Aline, Thouvenin Guillaume, Ullmann Nicola, Walker Woolf, Yiallouros Panayiotis, Kuehni Claudia E, Latzin Philipp, Beydon Nicole, Lucas Jane S
Аннотация
METHODS: Using a cross-sectional design, we prospectively collected clinical and concurrent microbiology data from 408 participants with probable or confirmed PCD, aged ≥5 years, from 12 countries. We used Global Lung Function Initiative 2012 references to calculate forced expiratory volume in 1 s (FEV1) z-scores. For 351 patients (86%) with complete data, we assessed the association of the four most frequently isolated pathogens with lung function by fitting multilevel linear models with country as random intercept, adjusted for age at diagnosis, age at lung function, use of antibiotic prophylaxis and body mass index z-scores.
RESULTS: Individuals with Pseudomonas aeruginosa growth in culture had significantly lower FEV1 z-scores (β= -0.87, 95% CI -1.40- -0.34), adjusted for presence of Haemophilus influenzae, methicillin-sensitive Staphylococcus aureus and Streptococcus pneumoniae, and for covariates. When stratified by age, associations remained strong for adults but not for children. Results were similar when ciliary defects by transmission electron microscopy were included in the models and when restricting analysis to only confirmed PCD cases.
CONCLUSIONS: We found that P. aeruginosa was associated with worse lung function in individuals with PCD, particularly adults. These findings suggest that it is prudent to aim for P. aeruginosa eradication in the first instance, and to treat exacerbations promptly in colonised patients.