Understanding primary ciliary dyskinesia

被引:0
|
作者
Ferkol, Thomas [1 ]
机构
[1] Univ N Carolina, Sch Med, Dept Pediat, Chapel Hill, NC USA
基金
美国国家卫生研究院;
关键词
airway; bronchiectasis; cilia; ciliopathies; heterotaxy; infection; inflammation; situs inversus totalis;
D O I
10.1002/ppul.27360
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Primary ciliary dyskinesia (PCD) is a rare, inherited disease characterized by impaired motile ciliary function leading to chronic sinopulmonary disease, persistent middle ear effusions, laterality defects, and subfertility. Over fifty PCD-associated genes have also been identified, which have provided new insights into the processes involved into ciliary assembly, structure, and function. Historically, the diagnosis of PCD was based on the presence of ultrastructural defects in the ciliary axoneme but with identification of a growing number of disease-associated genes, genetic testing has become a first-line diagnostic tool. Other approaches have also evolved, that have improved our diagnostic capabilities. Treatments for PCD have lagged, and though our growing understanding of the genetic and pathophysiological bases of the disease of PCD may yield to better therapeutic strategies.
引用
收藏
页数:2
相关论文
共 50 条
  • [41] PRIMARY CILIARY DYSKINESIA IN THE DOG
    MORRISON, WB
    WILSMAN, NJ
    FOX, LE
    FARNUM, CE
    JOURNAL OF VETERINARY INTERNAL MEDICINE, 1987, 1 (02) : 67 - 74
  • [42] Management of Primary Ciliary Dyskinesia
    Raidt, J.
    Brillault, J.
    Brinkmann, F.
    Jung, A.
    Koerner-Rettberg, C.
    Koitschev, A.
    Linz-Keul, H.
    Nuesslein, T.
    Ringshausen, F. C.
    Roehmel, J.
    Rosewich, M.
    Werner, C.
    Omran, H.
    PNEUMOLOGIE, 2020, 74 (11): : 750 - 765
  • [43] FAMILIAR PRIMARY CILIARY DYSKINESIA
    HUGBATSCHELET, H
    RUTISHAUSER, M
    HADZISELIMOVIC, F
    HELVETICA PAEDIATRICA ACTA, 1985, 40 (2-3) : 192 - 192
  • [44] Diagnosis of primary ciliary dyskinesia
    Kowal Olm, Mary Anne
    Caldini, Elia Garcia
    Mauad, Thais
    JORNAL BRASILEIRO DE PNEUMOLOGIA, 2015, 41 (03) : 251 - 263
  • [45] Primary ciliary dyskinesia (PCD)
    Meeks, M
    Bush, A
    PEDIATRIC PULMONOLOGY, 2000, 29 (04) : 307 - 316
  • [46] Alexithymia in primary ciliary dyskinesia
    Caglar, Hanife Tugce
    Pekcan, Sevgi
    Erden, Semih
    Unal, Gokcen
    Yilmaz, Asli Imran
    Ercan, Fatih
    Savas, Suat
    Ayman, Fatma Nur
    Istanbullu, Hasan Arif
    PEDIATRIC PULMONOLOGY, 2024, 59 (10) : 2457 - 2463
  • [47] Diagnostics of Primary Ciliary Dyskinesia
    Schrott-Fischer, A.
    Rieger, G.
    Morass, B.
    Bitsche, M.
    Horak, E.
    Riechelmann, H.
    Glueckert, R.
    LARYNGO-RHINO-OTOLOGIE, 2008, 87 (11) : 809 - 820
  • [48] Alexithymia in primary ciliary dyskinesia
    Caglar, Hanife Tugce
    Pekcan, Sevgi
    Ercan, Fatih
    Turkut, Asli
    Unal, Gokcen
    EUROPEAN RESPIRATORY JOURNAL, 2023, 62
  • [49] Diagnosing primary ciliary dyskinesia
    O'Callaghan, Christopher
    Chilvers, Mark
    Hogg, Claire
    Bush, Andrew
    Lucas, Jane
    THORAX, 2007, 62 (08) : 656 - 657
  • [50] Primary ciliary dyskinesia in adults
    Honore, I.
    Burgel, P. -R.
    REVUE DES MALADIES RESPIRATOIRES, 2016, 33 (02) : 165 - 189