Malena Cohen-Cymberknoh1, Nir Weigert2, Alex Gileles-Hillel3, Oded Breuer3, Natalia Simanovsky4, Mieke Boon5, Kris De Boeck5, Angelo Barbato6, Deborah Snijders6, Mirella Collura7, Ugo Pradal8, Hannah Blau9, Huda Mussaffi9, Mareike Price10, Lea Bentur11, Michal Gur11, Micha Aviram12, Elie Picard13, Michal Shteinberg14, Galit Livnat14, Joseph Rivlin14, Nurith Hiller4, David Shoseyov3, Israel Amirav15, Eitan Kerem3. 1. Pediatric Pulmonary Unit, Hadassah-Hebrew University Medical Center, Jerusalem, Israel. Electronic address: malena@hadassah.org.il. 2. Medical School, Hebrew University, Jerusalem, Israel. 3. Pediatric Pulmonary Unit, Hadassah-Hebrew University Medical Center, Jerusalem, Israel. 4. Department of Radiology, Hadassah-Hebrew University Medical Center, Jerusalem, Israel. 5. University Hospital Gasthuisberg, Leuven, Belgium. 6. Department of Pediatrics, University of Padova, Italy. 7. CRR Fibrosi Cistica and Department of Pediatrics, Ospedale Dei Bambini, G. Di Cristina, ARNAS Civico, Palermo, Italy. 8. CF Center, Azienda Ospedaliera di Verona, Italy. 9. Pediatric Pulmonary Unit, Schneider Children's Medical Center, Petach Tikva, Israel. 10. Children's University Hospital, Hannover, Germany. 11. Pediatric Pulmonary Unit, Ruth Rappaport Children's Hospital, Rambam Medical Center, Haifa, Israel. 12. Soroka Medical Center, Beer-Sheva, Israel. 13. Pediatric Pulmonary Unit, Shaare- Zedek Medical Center, Jerusalem, Israel. 14. Pulmonary Institute, Carmel Medical Center, Haifa, Israel. 15. Department of Pediatrics, University of Alberta, Canada.
Abstract
BACKGROUND: Airway infections in Primary Ciliary Dyskinesia (PCD) are caused by different microorganisms, including pseudomonas aeruginosa (PA). The aim of this study was to investigate the association of PA colonization and the progression of lung disease in PCD. METHODS: Data from 11PCD centers were retrospectively collected from 2008 to 2013. Patients were considered colonized if PA grew on at least two separate sputum cultures; otherwise, they were classified as non-colonized. These two groups were compared on the lung function computed tomography (CT) Brody score and other clinical parameters. RESULTS: Data were available from 217 patients; 60 (27.6%) of whom were assigned to the colonized group. Patients colonized with PA were older and were diagnosed at a later age. Baseline forced expiratory volume at 1 s (FEV1) was lower in the colonized group (72.4 ± 22.0 vs. 80.1 ± 18.9, % predicted, p = 0.015), but FEV1 declined throughout the study period was similar in both groups. The colonized group had significantly worse CT-Brody scores (36.07 ± 24.38 vs. 25.56 ± 24.2, p = 0.034). A subgroup analysis with more stringent definitions of colonization revealed similar results. CONCLUSIONS: Lung PA colonization in PCD is associated with more severe disease as shown by the FEV1 and CT score. However, the magnitude of decline in pulmonary function was similar in colonized and non-colonized PCD patients.
BACKGROUND:Airway infections in Primary Ciliary Dyskinesia (PCD) are caused by different microorganisms, including pseudomonas aeruginosa (PA). The aim of this study was to investigate the association of PA colonization and the progression of lung disease in PCD. METHODS: Data from 11PCD centers were retrospectively collected from 2008 to 2013. Patients were considered colonized if PA grew on at least two separate sputum cultures; otherwise, they were classified as non-colonized. These two groups were compared on the lung function computed tomography (CT) Brody score and other clinical parameters. RESULTS: Data were available from 217 patients; 60 (27.6%) of whom were assigned to the colonized group. Patients colonized with PA were older and were diagnosed at a later age. Baseline forced expiratory volume at 1 s (FEV1) was lower in the colonized group (72.4 ± 22.0 vs. 80.1 ± 18.9, % predicted, p = 0.015), but FEV1 declined throughout the study period was similar in both groups. The colonized group had significantly worse CT-Brody scores (36.07 ± 24.38 vs. 25.56 ± 24.2, p = 0.034). A subgroup analysis with more stringent definitions of colonization revealed similar results. CONCLUSIONS: Lung PA colonization in PCD is associated with more severe disease as shown by the FEV1 and CT score. However, the magnitude of decline in pulmonary function was similar in colonized and non-colonized PCDpatients.
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