From: Why, when and how to investigate primary ciliary dyskinesia in adult patients with bronchiectasis
Paper | Type and site of study | Patients involved | Patients screened for PCD | PCD diagnosed | Screening tests | Diagnosis tests | Features of PCD population |
---|---|---|---|---|---|---|---|
Amorim, 2015 [77] | Monocentric, Retrospective, Cohort study Portugal | 202 | 5 with history of infertility | 1 (0.5%) | Semen analysis | Not mentioned | Not mentioned |
Kadowaki, 2015 [78] | Monocentric, Retrospective, Cohort study Japan | 147 | 147 | 2 (1%) | Kartagener’s features | Kartagener’s features | Not mentioned |
Verra, 1991 [79] | Monocentric, Prospective, Cohort study France | 53 | 38 with diffuse bronchiectasis | 5 (13%) | Chest HRCT | TEM | Prevalence in North African patients 36% Prevalence in European patients 4% |
Pasteur, 2000 [80] | Monocentric, Prospective, Cohort study United Kingdom | 150 | 150 | 1 (0.6%) | Light microscopy with CBF | TEM | Not mentioned |
King, 2006 [81] | Monocentric, Prospective, Cross-sectional Australia | 103 | 103 | 1 (1%) | Unexplained infertility | Ciliary function | Not mentioned |
Shoemark, 2007 [11] | Monocentric, Prospective, Cohort study United Kingdom | 240 | 240 | 17 (10%) 12 PCD positive 5 PCD likely | Saccharin test nNO | Light microscopy TEM | Age 36 ± 13 |
Anwar, 2013 [82] | Two centres Prospective, Cohort study United Kingdom | 189 | 189 | 2 (1%) | History of upper and lower respiratory symptoms and/or infertility | Referral to specialized PCD centre | Not mentioned |
Qian, 2015 [83] | Multi-centre, Prospective, Cohort study China (Chinese Han ethnicity) | 476 | 71 with history of upper and lower respiratory symptoms or Kartagener’s features | 4 (0.9%) | Saccharin test | TEM | Not mentioned |
Lonni, 2015 [24] | Multicentre, International, Prospective, Cohort study Italy United Kingdom Spain Greece Belgium Ireland | 1258 | Not mentioned | 21 (1.7%) | History of upper and lower respiratory symptoms Saccharin test nNO | Referral to specialized PCD centre | Younger than 50 years Non-smokers Mild disease 11 (52.4%) Moderate 8 (38.1%) Severe 2 (9.5%) |
Guan, 2015 [84] | Multicentre, Prospective, Cohort study China | 148 | 148 | 2 (1.4%) | Saccharin test | Kartagener’s features | Younger than other bronchiectasis patients |
Dimakou, 2016 [85] | Monocentric, Prospective, Cohort study Greece | 277 | 32 with history of respiratory distress syndrome after birth, of upper and lower respiratory symptoms since childhood and/or infertility | 12 (4%) | Saccharin test nNO | TEM | Not mentioned |
Olveira, 2017 [86] | Multi-centre, Prospective, Cross-sectional Spain | 2047 | Not mentioned | 60 (2.9%) | Clinical features Saccharin test Seroalbumin NO (not specified if FENO or nNO) | TEM | Age 42.9 ± 8.8 Age at diagnosis 22.1 ± 18.1 Male 29 (48.3%) Smokers 14 (23.3%) BMI 23.8 ± 6.1 FEV1 (%) 67.1 ± 24.2 Chronic Infection Pa 27 (45%) Inhaled ATB 21 (39.6%) Bilateral bronchiectasis 29 (48.3%) |