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Spirometric indices in primary ciliary dyskinesia: systematic review and meta-analysis

Florian S. Halbeisen, Anu Jose, Carmen de Jong, Sylvia Nyilas, Philipp Latzin, Claudia E. Kuehni, Myrofora Goutaki
ERJ Open Research 2019 5: 00231-2018; DOI: 10.1183/23120541.00231-2018
Florian S. Halbeisen
1Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland
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  • ORCID record for Florian S. Halbeisen
Anu Jose
1Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland
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Carmen de Jong
1Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland
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Sylvia Nyilas
2Paediatric Respiratory Medicine, Children's University Hospital of Bern, University of Bern, Bern, Switzerland
3Dept of Diagnostic, Interventional and Pediatric Radiology, Inselspital, University of Bern, Bern, Switzerland
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Philipp Latzin
2Paediatric Respiratory Medicine, Children's University Hospital of Bern, University of Bern, Bern, Switzerland
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Claudia E. Kuehni
1Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland
2Paediatric Respiratory Medicine, Children's University Hospital of Bern, University of Bern, Bern, Switzerland
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Myrofora Goutaki
1Institute of Social and Preventive Medicine, University of Bern, Bern, Switzerland
2Paediatric Respiratory Medicine, Children's University Hospital of Bern, University of Bern, Bern, Switzerland
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  • ORCID record for Myrofora Goutaki
  • For correspondence: myrofora.goutaki@ispm.unibe.ch
  • Article
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Figures

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  • FIGURE 1
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    FIGURE 1

    Flowchart describing the selection procedure.

  • FIGURE 2
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    FIGURE 2

    Forced expiratory volume in 1 s (FEV1) in primary ciliary dyskinesia patients: forest plot showing the heterogeneity and weighted mean value of FEV1 % pred in the included studies.

  • FIGURE 3
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    FIGURE 3

    Forced expiratory volume in 1 s (FEV1) in primary ciliary dyskinesia patients by age group: forest plot showing the heterogeneity and weighted mean value of FEV1 % pred in the included studies.

  • FIGURE 4
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    FIGURE 4

    Forced vital capacity (FVC) in primary ciliary dyskinesia patients: forest plot showing the heterogeneity and weighted mean value of FVC % pred in the included studies.

Tables

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  • Supplementary Materials
  • TABLE 1

    Detailed characteristics of included studies describing spirometric indices of primary ciliary dyskinesia patients, stratified by age group of patients

    First author [ref.]CountryYear of publicationPatients nAge yearsType of studyStudy designDiagnosticsSymptoms stratified by ageLongitudinal dataFEV1FVC
    Children
     Al Saadi [51]Saudi Arabia20132210.86±3.09#ProspectiveCase–controlEM+HFVMNA−+−
     Davis [55]USA/Canada20151188 (5–11)¶ProspectiveCase seriesEM or genetic analysisNA−+−
     Gokdemir [58]Turkey20142412.9±2.7#ProspectiveClinical trialEM or KartagenerNA−++
     Karadag [61]UK19992110.8±3.2#RetrospectiveCase–controlEM, HFVMNA−++
     Koh [62]Korea20001912 (7–16)+ProspectiveClinical trialEMNA−+−
     Maglione [63]Denmark, Italy and UK20141588.7 (4.2–17.4)§RetrospectiveCohortAccording to consensusNA+++
     Paff [68]The Netherlands20132510.7 (7.1–14.5)¶ProspectiveCase–controlAccording to consensusNA−+−
    Adults
     Frija-Masson [57]France20177834.8 (28.6–47.1)¶RetrospectiveCase seriesEM, genetic analysis or KartagenerNA+++
     Horvath [60]UK20031435±4.6#ProspectiveCase–controlEM, HFVMNA−++
     Mossberg [65]Sweden19832430 (19–47)§ProspectiveCase seriesClinical suspicionNA−+−
    Both children and adults
     Amirav [52]Israel20161310 (5–43)¶RetrospectiveCase seriesAll tests, including genetic analysis−−+−
     Boon [53]Belgium201412217.7 (9.5–28.1)¶RetrospectiveCase seriesEM, HFVM, genetic analysis−−++
     Cohen-Cymberknoh [54]Israel20141415.9±8.6#RetrospectiveCase seriesEM, HFVM, nNO, genetic analysis−−+−
     Ellerman [56]Denmark19972421±13.6#ProspectiveCase seriesHFVM++++
     Hellinckx [59]Belgium19981115.2±7#ProspectiveCase seriesEM, HFVM+++−
     Mahut [64]France20061613.5 (12–17)¶RetrospectiveCase seriesClinical, EM+−+−
     Noone [4]USA and Canada200478Adults: 36 (19–73)+; children: 8 (1–17)+ProspectiveCase seriesEM, HFVM, genetic analysis+−+−
     Nyilas [66]Germany and Switzerland20174914.7±6.6#ProspectiveCase–controlAll tests−−++
     Olm [67]Brazil20111013.4±4.7#ProspectiveCase–controlEM+HFVM+−++
     Pifferi [69]Italy20154514 (22.25)¶ProspectiveCase–controlEM, HFVM, nNO−−++
     Shah [70]UK201611823.5 (10–36)¶RetrospectiveCase seriesEM, HFVM, nNO, 2% clinical−++−
     Tolusakow [71]Germany1981213–43ƒRetrospectiveCase seriesClinical suspicion−−++
     Vallet [72]France2013416 (3–11)¶RetrospectiveCase seriesEM, HFVM−+++
     Yiallouros [73]Cyprus2015200.1–58.4ƒRetrospectiveCase seriesEM or HFVM+nNO++++

    FEV1: forced expiratory volume in 1 s; FVC: forced vital capacity; EM: electron microscopy; HFVM: high-frequency video-microscopy; NA: not applicable; nNO: nasal nitric oxide. Age is reported in different formats depending on the available information: #: mean±sd; ¶: median (interquartile range); +: median (range); §: mean (range); ƒ: range.

    • TABLE 2

      Characteristics of included studies describing spirometry indices of primary ciliary dyskinesia patients

      Total studies24 (100)
      Study size
       ≤20 patients8 (33)
       21–50 patients10 (42)
       51–100 patients2 (8)
       >100 patients4 (17)
      Number of centres
       Single centre17 (71)
       Multicentre7 (29)
      Publication period
       ≤19962 (8)
       1997–20077 (29)
       ≥200815 (63)
      Study region
       Europe15 (62.5)
       Asia6 (25)
       North and South America3 (12.5)
      Age of participants
       Children <18 years7 (29)
       Adults ≥18 years3 (13)
       Children and adults14 (58)
      Study design
       Retrospective11 (46)
       Prospective13 (54)
      Diagnostics performed
       Diagnosis based only on strong clinical suspicion2 (8)
       Diagnosis based on EM or a combination of tests in most patients; few only on strong clinical suspicion#3 (13)
       Diagnosis based on EM or a combination of tests in all patients#19 (79)
      Spirometry indices reported
       FEV124 (100)
       FVC12 (50)
      Information for different age groups6 (43)¶
      Longitudinal information8 (33)

      Data are presented as n (%). EM: electron microscopy; FEV1: forced expiratory volume in 1 s; FVC: forced vital capacity. #: combination of the following: EM, nasal nitric oxide, high-frequency video-microscopy or light microscopy, or genetic analysis. ¶: of 14 studies including children and adults.

      Supplementary Materials

      • Figures
      • Tables
      • Supplementary Material

        Please note: supplementary material is not edited by the Editorial Office, and is uploaded as it has been supplied by the author.

        TABLE S1 Characteristics of included studies describing spirometric indices of PCD patients by country, including studies with overlapping population 00231-2018.tableS1

        TABLE S2 Metaregression results on spirometric indices (FEV1 and FVC) of PCD patients 00231-2018.tableS2

        FIGURE S1 Forest plot showing the heterogeneity and weighted mean value of FVC percent predicted in the included publications 00231-2018.figureS1

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      Spirometric indices in primary ciliary dyskinesia: systematic review and meta-analysis
      Florian S. Halbeisen, Anu Jose, Carmen de Jong, Sylvia Nyilas, Philipp Latzin, Claudia E. Kuehni, Myrofora Goutaki
      ERJ Open Research Apr 2019, 5 (2) 00231-2018; DOI: 10.1183/23120541.00231-2018

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      Spirometric indices in primary ciliary dyskinesia: systematic review and meta-analysis
      Florian S. Halbeisen, Anu Jose, Carmen de Jong, Sylvia Nyilas, Philipp Latzin, Claudia E. Kuehni, Myrofora Goutaki
      ERJ Open Research Apr 2019, 5 (2) 00231-2018; DOI: 10.1183/23120541.00231-2018
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