Literature DB >> 28877972

Clinical care for primary ciliary dyskinesia: current challenges and future directions.

Bruna Rubbo1,2, Jane S Lucas3,2.   

Abstract

Primary ciliary dyskinesia (PCD) is a rare genetic disease that affects the motility of cilia, leading to impaired mucociliary clearance. It is estimated that the vast majority of patients with PCD have not been diagnosed as such, providing a major obstacle to delivering appropriate care. Challenges in diagnosing PCD include lack of disease-specific symptoms and absence of a single, "gold standard", diagnostic test. Management of patients is currently not based on high-level evidence because research findings are mostly derived from small observational studies with limited follow-up period. In this review, we provide a critical overview of the available literature on clinical care for PCD patients, including recent advances. We identify barriers to PCD research and make suggestions for overcoming challenges.
Copyright ©ERS 2017.

Entities:  

Mesh:

Year:  2017        PMID: 28877972     DOI: 10.1183/16000617.0023-2017

Source DB:  PubMed          Journal:  Eur Respir Rev        ISSN: 0905-9180


  14 in total

1.  Bi-allelic BRWD1 variants cause male infertility with asthenoteratozoospermia and likely primary ciliary dyskinesia.

Authors:  Ting Guo; Chao-Feng Tu; Dan-Hui Yang; Shui-Zi Ding; Cheng Lei; Rong-Chun Wang; Lv Liu; Xi Kang; Xiao-Qing Shen; Yi-Feng Yang; Zhi-Ping Tan; Yue-Qiu Tan; Hong Luo
Journal:  Hum Genet       Date:  2021-01-03       Impact factor: 4.132

2.  Rab35 controls cilium length, function and membrane composition.

Authors:  Stefanie Kuhns; Cecília Seixas; Sara Pestana; Bárbara Tavares; Renata Nogueira; Raquel Jacinto; José S Ramalho; Jeremy C Simpson; Jens S Andersen; Arnaud Echard; Susana S Lopes; Duarte C Barral; Oliver E Blacque
Journal:  EMBO Rep       Date:  2019-08-21       Impact factor: 8.807

3.  CiliarMove: new software for evaluating ciliary beat frequency helps find novel mutations by a Portuguese multidisciplinary team on primary ciliary dyskinesia.

Authors:  Pedro Sampaio; Mónica Ferro da Silva; Inês Vale; Mónica Roxo-Rosa; Andreia Pinto; Carolina Constant; Luisa Pereira; Carla M Quintão; Susana S Lopes
Journal:  ERJ Open Res       Date:  2021-02-08

4.  Proceedings of the 4th BEAT-PCD Conference and 5th PCD Training School.

Authors:  Laura E Gardner; Katie L Horton; Amelia Shoemark; Jane S Lucas; Kim G Nielsen; Helene Kobbernagel; Bruna Rubbo; Robert A Hirst; Panayiotis Kouis; Nicola Ullmann; Ana Reula; Nisreen Rumman; Hannah M Mitchison; Andreia Pinto; Charlotte Richardson; Anne Schmidt; James Thompson; René Gaupmann; Maciej Dabrowski; Pleasantine Mill; Siobhan B Carr; Dominic P Norris; Claudia E Kuehni; Myrofora Goutaki; Claire Hogg
Journal:  BMC Proc       Date:  2020-06-19

5.  Primary Ciliary Dyskinesia as a Cause of Repeating Atelectasis in the Neonatal Period.

Authors:  Arlete Esteves Lopes Primo; Romy Schmidt Brock Zacharias; Amanda Dias de Moraes; Luiz Vicente Ribeiro Ferreira da Silva Filho; Daniela Rodriguez Trujillo; Renata de Araújo Monteiro Yoshida; Arno Noberto Warth; Celso Moura Rebello
Journal:  Am J Case Rep       Date:  2020-06-11

6.  Pulmonary exacerbations in patients with primary ciliary dyskinesia: an expert consensus definition for use in clinical trials.

Authors:  Jane S Lucas; Florian Gahleitner; Adelina Amorim; Mieke Boon; Philippa Brown; Carolina Constant; Simon Cook; Suzanne Crowley; Damien M S Destouches; Ernst Eber; Huda Mussaffi; Eric Haarman; Amanda Harris; Cordula Koerner-Rettberg; Claudia E Kuehni; Philipp Latzin; Michael R Loebinger; Natalie Lorent; Bernard Maitre; Antonio Moreno-Galdó; Kim G Nielsen; Uğur Özçelik; Lue Katrine Drasbæk Philipsen; Petr Pohunek; Eva Polverino; Jessica Rademacher; Phil Robinson; Deborah Snijders; Panayiotis Yiallouros; Siobhán B Carr
Journal:  ERJ Open Res       Date:  2019-02-01

7.  Clinical and Genetic Analysis of Children with Kartagener Syndrome.

Authors:  Rute Pereira; Telma Barbosa; Luís Gales; Elsa Oliveira; Rosário Santos; Jorge Oliveira; Mário Sousa
Journal:  Cells       Date:  2019-08-15       Impact factor: 6.600

8.  Brazilian consensus on non-cystic fibrosis bronchiectasis.

Authors:  Mônica Corso Pereira; Rodrigo Abensur Athanazio; Paulo de Tarso Roth Dalcin; Mara Rúbia Fernandes de Figueiredo; Mauro Gomes; Clarice Guimarães de Freitas; Fernando Ludgren; Ilma Aparecida Paschoal; Samia Zahi Rached; Rosemeri Maurici
Journal:  J Bras Pneumol       Date:  2019-08-12       Impact factor: 2.624

Review 9.  Sperm defects in primary ciliary dyskinesia and related causes of male infertility.

Authors:  Anu Sironen; Amelia Shoemark; Mitali Patel; Michael R Loebinger; Hannah M Mitchison
Journal:  Cell Mol Life Sci       Date:  2019-11-28       Impact factor: 9.261

10.  Mutations in PIH proteins MOT48, TWI1 and PF13 define common and unique steps for preassembly of each, different ciliary dynein.

Authors:  Ryosuke Yamamoto; Shiho Yanagi; Masahito Nagao; Yuya Yamasaki; Yui Tanaka; Winfield S Sale; Toshiki Yagi; Takahide Kon
Journal:  PLoS Genet       Date:  2020-11-03       Impact factor: 5.917

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