Please use this identifier to cite or link to this item: https://covid-19.conacyt.mx/jspui/handle/1000/384
Late diagnosis of infants with PCD and neonatal respiratory distress
Chris O Callaghan
Ugur Ozcelik
Sergio Scigliano
Santiago Ucros
Panayiotis Yiallouros
Sven M Schulzke
Angelo Barbato
Suzanne Crowley
Amanda Harris
Robert A Hirst
Bulent Karadag
Vendula Martinu
Lucy Morgan
Myrofora Goutaki
Claudia E Kuehni
Florian S Halbeisen
Novel Coronavirus
Acceso Abierto
Atribución-NoComercial-SinDerivadas
10.1101/2020.03.27.20045377
Neonatal respiratory distress (NRD) is common among infants with primary ciliary dyskinesia (PCD), but we do not know whether affected neonates are diagnosed timely. We used data from the international PCD cohort study (iPCD), including only participants diagnosed between 2000 and 2019 using current diagnostic criteria. We assessed the proportion of patients with PCD with a history of NRD and their age at diagnosis, stratifying by presence of laterality defects. First we analysed data from the entire cohort and then from a subgroup including children diagnosed using stricter criteria. Among the 1375 patients in the study, 45% had a history of NRD and 42% a laterality defect. Out of the 476 children with definite PCD diagnosis, 55% had a history of NRD and 50% a laterality defect. PCD was diagnosed at a median age of 3.4 years in this group, varying from less than 1 year in Norway and Cyprus to 10 years in Turkey. Overall, 30% of children with PCD were diagnosed during the first 12 months of life. This varied from 13% in those with situs solitus and no NRD, to 21% in those with situs solitus and NRD, 33% in those with situs anomalies but no NRD, and 52% in those with both NRD and situs anomalies. Our results suggest that we need to improve our knowledge of the neonatal presentation of infants with PCD, and apply this knowledge in neonatology so that these patients will receive appropriate care sooner, at the start of their extrauterine life. ### Competing Interest Statement The authors have declared no competing interest. ### Clinical Trial NCT03517865 ### Funding Statement This study was supported by the Swiss National Foundation (SNF 320030_173044). The setting up of the iPCD Cohort (salaries, consumables, and equipment) was funded by the EU FP7 project BESTCILIA (http://bestcilia.eu) and several Swiss funding bodies including the Lung Leagues of Bern, St Gallen, Vaud, Ticino, and Valais, and the Milena Carvajal Pro-Kartagener Foundation. Data collection and management at each site was funded according to local arrangements. Most participating researchers and data contributors participate in the COST Action BEAT-PCD: Better Evidence to Advance Therapeutic options for PCD (BM 1407, www.beatpcd.org) and the ERN-LUNG (PCD core). The Czech cohort data collection has been supported by the grant No. NV19-07-00210 by the Ministry of Health, Czech Republic. . PCD research at the University of Leicester is supported by the NIHR GOSH BRC. The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health. ### Author Declarations All relevant ethical guidelines have been followed; any necessary IRB and/or ethics committee approvals have been obtained and details of the IRB/oversight body are included in the manuscript. Yes All necessary patient/participant consent has been obtained and the appropriate institutional forms have been archived. Yes I understand that all clinical trials and any other prospective interventional studies must be registered with an ICMJE-approved registry, such as ClinicalTrials.gov. I confirm that any such study reported in the manuscript has been registered and the trial registration ID is provided (note: if posting a prospective study registered retrospectively, please provide a statement in the trial ID field explaining why the study was not registered in advance). Yes I have followed all appropriate research reporting guidelines and uploaded the relevant EQUATOR Network research reporting checklist(s) and other pertinent material as supplementary files, if applicable. Yes The ethical approval of the study does not allow to have the datasetopenly available. Data are available to researchers upon request by the study PIs
Cold Spring Harbor Laboratory Press
2020
Preimpreso
https://www.medrxiv.org/content/10.1101/2020.03.27.20045377v1
Inglés
VIRUS RESPIRATORIOS
Appears in Collections:Artículos científicos

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