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Critical Evaluation of Sinonasal Disease in 64 Adults with Primary Ciliary Dyskinesia

To date, no study precisely described ear, nose and throat (ENT) disease in adults with primary ciliary dyskinesia (PCD) and its relationship with ciliary function/ultrastructure. A retrospective study of standardized ENT data (exam, audiogram, sinus Computed tomography (CT), and bacteriology) was c...

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Autores principales: Bequignon, Emilie, Dupuy, Laurence, Zerah-Lancner, Francoise, Bassinet, Laurence, Honoré, Isabelle, Legendre, Marie, Devars du Mayne, Marie, Escabasse, Virginie, Crestani, Bruno, Maître, Bernard, Escudier, Estelle, Coste, André, Papon, Jean-François
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6571605/
https://www.ncbi.nlm.nih.gov/pubmed/31067752
http://dx.doi.org/10.3390/jcm8050619
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author Bequignon, Emilie
Dupuy, Laurence
Zerah-Lancner, Francoise
Bassinet, Laurence
Honoré, Isabelle
Legendre, Marie
Devars du Mayne, Marie
Escabasse, Virginie
Crestani, Bruno
Maître, Bernard
Escudier, Estelle
Coste, André
Papon, Jean-François
author_facet Bequignon, Emilie
Dupuy, Laurence
Zerah-Lancner, Francoise
Bassinet, Laurence
Honoré, Isabelle
Legendre, Marie
Devars du Mayne, Marie
Escabasse, Virginie
Crestani, Bruno
Maître, Bernard
Escudier, Estelle
Coste, André
Papon, Jean-François
author_sort Bequignon, Emilie
collection PubMed
description To date, no study precisely described ear, nose and throat (ENT) disease in adults with primary ciliary dyskinesia (PCD) and its relationship with ciliary function/ultrastructure. A retrospective study of standardized ENT data (exam, audiogram, sinus Computed tomography (CT), and bacteriology) was conducted in 64 adults with confirmed PCD who were followed in two ENT reference centers. Rhinorrhoea and hearing loss were the main symptoms. Symptom scores were higher in older patients. Nasal endoscopy was abnormal in all patients except one, showing nasal polyps in one-third of the patients and stagnant nasal mucus secretions in 87.5% of the patients. Sinus CT opacities were mainly incomplete and showed one-third of the patients with sinus hypoplasia and/or agenesis. Middle meatus mainly grew Haemophilus influenzae, Streptoccocus pneumoniae and Pseudomonas aeruginosa. Otitis media with effusion (OME), which is constant in childhood, was diagnosed in less than one-quarter of the patients. In two-thirds of the patients, audiogram showed hearing loss that was sensorineural in half of the patients. ENT disease severity was not correlated with ciliary function and ultrastructure, but the presence of OME was significantly associated with a forced expiratory volume (FEV1) < 70%. Rhinosinusitis is the most common clinical feature of PCD in adults, while OME is less frequent. The presence of active OME in adults with PCD could be a severity marker of lung function and lead to closer monitoring.
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spelling pubmed-65716052019-06-18 Critical Evaluation of Sinonasal Disease in 64 Adults with Primary Ciliary Dyskinesia Bequignon, Emilie Dupuy, Laurence Zerah-Lancner, Francoise Bassinet, Laurence Honoré, Isabelle Legendre, Marie Devars du Mayne, Marie Escabasse, Virginie Crestani, Bruno Maître, Bernard Escudier, Estelle Coste, André Papon, Jean-François J Clin Med Article To date, no study precisely described ear, nose and throat (ENT) disease in adults with primary ciliary dyskinesia (PCD) and its relationship with ciliary function/ultrastructure. A retrospective study of standardized ENT data (exam, audiogram, sinus Computed tomography (CT), and bacteriology) was conducted in 64 adults with confirmed PCD who were followed in two ENT reference centers. Rhinorrhoea and hearing loss were the main symptoms. Symptom scores were higher in older patients. Nasal endoscopy was abnormal in all patients except one, showing nasal polyps in one-third of the patients and stagnant nasal mucus secretions in 87.5% of the patients. Sinus CT opacities were mainly incomplete and showed one-third of the patients with sinus hypoplasia and/or agenesis. Middle meatus mainly grew Haemophilus influenzae, Streptoccocus pneumoniae and Pseudomonas aeruginosa. Otitis media with effusion (OME), which is constant in childhood, was diagnosed in less than one-quarter of the patients. In two-thirds of the patients, audiogram showed hearing loss that was sensorineural in half of the patients. ENT disease severity was not correlated with ciliary function and ultrastructure, but the presence of OME was significantly associated with a forced expiratory volume (FEV1) < 70%. Rhinosinusitis is the most common clinical feature of PCD in adults, while OME is less frequent. The presence of active OME in adults with PCD could be a severity marker of lung function and lead to closer monitoring. MDPI 2019-05-07 /pmc/articles/PMC6571605/ /pubmed/31067752 http://dx.doi.org/10.3390/jcm8050619 Text en © 2019 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Bequignon, Emilie
Dupuy, Laurence
Zerah-Lancner, Francoise
Bassinet, Laurence
Honoré, Isabelle
Legendre, Marie
Devars du Mayne, Marie
Escabasse, Virginie
Crestani, Bruno
Maître, Bernard
Escudier, Estelle
Coste, André
Papon, Jean-François
Critical Evaluation of Sinonasal Disease in 64 Adults with Primary Ciliary Dyskinesia
title Critical Evaluation of Sinonasal Disease in 64 Adults with Primary Ciliary Dyskinesia
title_full Critical Evaluation of Sinonasal Disease in 64 Adults with Primary Ciliary Dyskinesia
title_fullStr Critical Evaluation of Sinonasal Disease in 64 Adults with Primary Ciliary Dyskinesia
title_full_unstemmed Critical Evaluation of Sinonasal Disease in 64 Adults with Primary Ciliary Dyskinesia
title_short Critical Evaluation of Sinonasal Disease in 64 Adults with Primary Ciliary Dyskinesia
title_sort critical evaluation of sinonasal disease in 64 adults with primary ciliary dyskinesia
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6571605/
https://www.ncbi.nlm.nih.gov/pubmed/31067752
http://dx.doi.org/10.3390/jcm8050619
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