Lysosomal storage disorders (LSDs) are a diverse group of inborn errors of metabolism, which involves the storage of macromolecules within the lysosome due to the absence of or deficiency in lysosomal enzymes. In this condition, respiratory dysfunction is the common cause of morbidity and mortality. Thus, an objective measurement examination of pulmonary function, such as spirometry, is essential for the diagnosis and management of respiratory conditions. However, spirometry requires active patient involvement. Meanwhile, the forced oscillation technique (FOT) is performed during tidal breathing and is feasible in patients who cannot perform spirometry. Thus, the current study aimed to assess the feasibility and efficacy of FOT in assessing respiratory function among patients with LSD. This study was conducted at the outpatient department of Tawam Hospital, Al-Ain, the United Arab Emirates. Patients with confirmed LSD were included in the analysis. Information on respiratory history was obtained using a standardized respiratory questionnaire, and lung function was assessed using FOT. The main outcomes were respiratory resistance (Rrs5), reactance at 5 Hz (Xrs5), and area under the reactance curve (AX). In total, 35 patients with LSD were assessed. FOT was used to evaluate 16 (47%) patients. Of the patients who were successfully assessed using FOT, 10 presented with abnormal outcomes. Hence, there was an association between lung function assessed using FOT and spirometry results in these patients. FOT can help obtain lung function measurements in patients with LSD. In this study, the patients commonly presented with abnormal FOT outcomes.
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http://dx.doi.org/10.1038/s41598-025-92076-8 | DOI Listing |
Sci Rep
February 2025
Department of Genetics and Genomics, College of Medicine and Health Sciences, United Arab Emirates University, Al Ain, United Arab Emirates.
Lysosomal storage disorders (LSDs) are a diverse group of inborn errors of metabolism, which involves the storage of macromolecules within the lysosome due to the absence of or deficiency in lysosomal enzymes. In this condition, respiratory dysfunction is the common cause of morbidity and mortality. Thus, an objective measurement examination of pulmonary function, such as spirometry, is essential for the diagnosis and management of respiratory conditions.
View Article and Find Full Text PDFAllergy
February 2025
National Heart & Lung Institute, Imperial College London, London, UK.
Background: Post hoc analysis of clinical trials shows blood eosinophil counts (BEC) predict future preschool wheeze attacks; however, prospective usefulness in a clinical setting is unreported. We assessed the feasibility of point-of-care (POC) eosinophil measurements in preschool wheezers and related BEC to symptoms, lung function, and utility in predicting attacks.
Methods: Children aged 1-5 years with recurrent wheeze underwent finger-prick sampling during the outpatient clinic for POC eosinophils, forced oscillation technique (FOT) and/or spirometry, and symptom score (TRACK questionnaire).
Cureus
December 2024
Physiology, All India Institute of Medical Sciences, Nagpur, Nagpur, IND.
Introduction: Smoking remains a significant public health concern, associated with a myriad of diseases, including chronic obstructive pulmonary disease, cardiovascular diseases, and various cancers. While spirometry is the gold standard for assessing lung function, it may lack sensitivity in detecting early changes in respiratory mechanics induced by smoking. This study aims to evaluate the clinical potential of the forced oscillation technique (FOT) in detecting early changes in the respiratory mechanics of smokers and its correlation with computerized spirometry.
View Article and Find Full Text PDFMuscle Nerve
March 2025
Division of Pediatric Cardiology, Vanderbilt University Medical Center, Nashville, Tennessee, US.
Introduction/aims: Skeletal muscle magnetic resonance imaging (MRI) is a validated noninvasive tool to assess Duchenne muscular dystrophy (DMD) progression. There is interest in finding DMD biomarkers that decrease the burden of clinical trial participation, such as wearable devices. Our aim was to evaluate the relationship between activity, via accelerometry, and skeletal muscle MRI, particularly T mapping.
View Article and Find Full Text PDFCureus
November 2024
Department of Orthodontics and Dentofacial Orthopedics, Kothiwal Dental College and Research Centre, Moradabad, IND.
Introduction Gingival enlargement (GE) poses a significant problem during fixed orthodontic treatment (FOT). Thus, the primary aim of the current study was to evaluate the concentrations of biomarkers present in the gingival crevicular fluid (GCF) of individuals receiving FOT. The ancillary aim was to examine and compare biomarker levels among patients exhibiting GE undergoing FOT, those without GE undergoing FOT, and a control group comprising individuals not undergoing FOT and to assess the predictors for GE in patients undergoing orthodontic treatment.
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