Respiratory phenotypes of neuromuscular diseases: A challenging issue for pediatricians

Neuromuscular disease (NMDs) encompass a heterogeneous group of genetic disorders, with respiratory problems of variable intensity and progression described at any pediatric age, from infancy to adolescence, and they are largely associated with significant lifelong morbidity and high mortality. Rest...

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Main Authors: Melissa Borrelli, Gaetano Terrone, Roberto Evangelisti, Flora Fedele, Adele Corcione, Francesca Santamaria
Format: Article
Language:English
Published: Elsevier 2023-03-01
Series:Pediatrics and Neonatology
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Online Access:http://www.sciencedirect.com/science/article/pii/S1875957222002728
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author Melissa Borrelli
Gaetano Terrone
Roberto Evangelisti
Flora Fedele
Adele Corcione
Francesca Santamaria
author_facet Melissa Borrelli
Gaetano Terrone
Roberto Evangelisti
Flora Fedele
Adele Corcione
Francesca Santamaria
author_sort Melissa Borrelli
collection DOAJ
description Neuromuscular disease (NMDs) encompass a heterogeneous group of genetic disorders, with respiratory problems of variable intensity and progression described at any pediatric age, from infancy to adolescence, and they are largely associated with significant lifelong morbidity and high mortality. Restriction of breathing, impaired gas exchange, decline of lung function and sleep disordered breathing progressively develop because of muscular weakness and culminate in respiratory failure. Depending on the disease progression, airways manifestations can take weeks to months or even years to evolve, thus depicting two major respiratory phenotypes, characterized by rapid or slow progression to respiratory failure. Assessing type and age at onset of airways problems and their evolution over time can support pediatricians in the diagnostic assessment of NMD. In addition, knowing the characteristics of patients' respiratory phenotype can increase the level of awareness among neonatologists, geneticists, neurologists, pulmonologists, nutritionists, and chest therapists, supporting them in the challenging task of the multidisciplinary medical care of patients. In this review we examine the issues related to the pediatric respiratory phenotypes of NMD and present a novel algorithm that can act as a guide for the diagnostic agenda and the key preventive or therapeutic interventions of airways manifestations. With prolonged survival of children with NMD, the advent of neuromuscular respiratory medicine, including accurate assessment of the respiratory phenotype, will help physicians to determine patients’ prognoses and to design studies for the evaluation of new therapies.
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spelling doaj-art-f4bdfea8bf7840e3a60f5a7f4e9ae9f12025-08-20T02:22:10ZengElsevierPediatrics and Neonatology1875-95722023-03-0164210911810.1016/j.pedneo.2022.09.016Respiratory phenotypes of neuromuscular diseases: A challenging issue for pediatriciansMelissa Borrelli0Gaetano Terrone1Roberto Evangelisti2Flora Fedele3Adele Corcione4Francesca Santamaria5Department of Translational Medical Sciences, Pediatric Pulmonology, ItalyDepartment of Translational Medical Sciences, Child Neuropsychiatry Units, Federico II University, Naples, ItalyDepartment of Translational Medical Sciences, Pediatric Pulmonology, ItalyDepartment of Translational Medical Sciences, Pediatric Pulmonology, ItalyDepartment of Translational Medical Sciences, Pediatric Pulmonology, ItalyDepartment of Translational Medical Sciences, Pediatric Pulmonology, Italy; Corresponding author. Federico II University, Naples, Italy.Neuromuscular disease (NMDs) encompass a heterogeneous group of genetic disorders, with respiratory problems of variable intensity and progression described at any pediatric age, from infancy to adolescence, and they are largely associated with significant lifelong morbidity and high mortality. Restriction of breathing, impaired gas exchange, decline of lung function and sleep disordered breathing progressively develop because of muscular weakness and culminate in respiratory failure. Depending on the disease progression, airways manifestations can take weeks to months or even years to evolve, thus depicting two major respiratory phenotypes, characterized by rapid or slow progression to respiratory failure. Assessing type and age at onset of airways problems and their evolution over time can support pediatricians in the diagnostic assessment of NMD. In addition, knowing the characteristics of patients' respiratory phenotype can increase the level of awareness among neonatologists, geneticists, neurologists, pulmonologists, nutritionists, and chest therapists, supporting them in the challenging task of the multidisciplinary medical care of patients. In this review we examine the issues related to the pediatric respiratory phenotypes of NMD and present a novel algorithm that can act as a guide for the diagnostic agenda and the key preventive or therapeutic interventions of airways manifestations. With prolonged survival of children with NMD, the advent of neuromuscular respiratory medicine, including accurate assessment of the respiratory phenotype, will help physicians to determine patients’ prognoses and to design studies for the evaluation of new therapies.http://www.sciencedirect.com/science/article/pii/S1875957222002728neuromuscular diseasesrespiratory failurerespiratory muscles weaknessrespiratory phenotypesleepdisordered breathing
spellingShingle Melissa Borrelli
Gaetano Terrone
Roberto Evangelisti
Flora Fedele
Adele Corcione
Francesca Santamaria
Respiratory phenotypes of neuromuscular diseases: A challenging issue for pediatricians
Pediatrics and Neonatology
neuromuscular diseases
respiratory failure
respiratory muscles weakness
respiratory phenotype
sleepdisordered breathing
title Respiratory phenotypes of neuromuscular diseases: A challenging issue for pediatricians
title_full Respiratory phenotypes of neuromuscular diseases: A challenging issue for pediatricians
title_fullStr Respiratory phenotypes of neuromuscular diseases: A challenging issue for pediatricians
title_full_unstemmed Respiratory phenotypes of neuromuscular diseases: A challenging issue for pediatricians
title_short Respiratory phenotypes of neuromuscular diseases: A challenging issue for pediatricians
title_sort respiratory phenotypes of neuromuscular diseases a challenging issue for pediatricians
topic neuromuscular diseases
respiratory failure
respiratory muscles weakness
respiratory phenotype
sleepdisordered breathing
url http://www.sciencedirect.com/science/article/pii/S1875957222002728
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