Your browser doesn't support javascript.
loading
Show: 20 | 50 | 100
Results 1 - 1 de 1
Filter
Add more filters











Database
Language
Publication year range
1.
Sci Rep ; 9(1): 8693, 2019 06 18.
Article in English | MEDLINE | ID: mdl-31213628

ABSTRACT

Primary Ciliary Dyskinesia (PCD) is underdiagnosed in Brazil. We enrolled patients from an adult service of Bronchiectasis over a two-year period in a cross-sectional study. The inclusion criteria were laterality disorders (LD), cough with recurrent infections and the exclusion of other causes of bronchiectasis. Patients underwent at least two of the following tests: nasal nitric oxide, ciliary movement and analysis of ciliary immunofluorescence, and genetic tests (31 PCD genes + CFTR gene). The clinical characterization included the PICADAR and bronchiectasis scores, pulmonary function, chronic Pseudomonas aeruginosa (cPA) colonization, exhaled breath condensate (EBC) and mucus rheology (MR). Forty-nine of the 500 patients were diagnosed with definite (42/49), probable (5/49), and clinical (2/49) PCD. Twenty-four patients (24/47) presented bi-allelic pathogenic variants in a total of 31 screened PCD genes. A PICADAR score > 5 was found in 37/49 patients, consanguinity in 27/49, LD in 28/49, and eight PCD sibling groups. FACED diagnosed 23/49 patients with moderate or severe bronchiectasis; FEV1 ≤ 50% in 25/49 patients, eight patients had undergone lung transplantation, four had been lobectomized and cPA+ was determined in 20/49. The EBC and MR were altered in all patients. This adult PCD population was characterized by consanguinity, severe lung impairment, genetic variability, altered EBC and MR.


Subject(s)
Kartagener Syndrome/diagnosis , Lung Diseases/diagnosis , Pseudomonas Infections/diagnosis , Adult , Aged , Brazil/epidemiology , Comorbidity , Cross-Sectional Studies , Female , Genetic Testing , Humans , Kartagener Syndrome/epidemiology , Kartagener Syndrome/genetics , Lung Diseases/epidemiology , Lung Diseases/genetics , Male , Middle Aged , Prevalence , Pseudomonas Infections/epidemiology , Pseudomonas Infections/microbiology , Pseudomonas aeruginosa/isolation & purification , Pseudomonas aeruginosa/physiology , Severity of Illness Index , Young Adult
SELECTION OF CITATIONS
SEARCH DETAIL