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Dublin Core
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Title
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Treatment of Symptoms in Children with Q3 Conditions Scoping Review Results
Text
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URL Address
<a href="http://doi.org/10.1016/S0165-5876(01)00417-7" target="_blank" rel="noreferrer noopener">http://doi.org/​10.1016/S0165-5876(01)00417-7</a>
Dublin Core
The Dublin Core metadata element set is common to all Omeka records, including items, files, and collections. For more information see, http://dublincore.org/documents/dces/.
Title
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Disordered breathing during sleep in patients with mucopolysaccharidoses
Publisher
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International Journal of Pediatric Otorhinolaryngology
Date
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2001
Subject
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breathing difficulties; sleep disturbance; Galactosialidosis; MPSI; MPSII; MPSIII; MPSIIIA; MPSIIIB; MPSVI; MPSVII; trajectory; characteristics; obstructive sleep apnea; OSA; frequent awakenings; poor sleep quality
Creator
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Leighton SEJ; Papsin B; Vellodi A; Dinwiddie R; Lane R
Description
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Objective: Obstructive sleep apnoea (OSA) has been reported as a feature of children with mucopolysaccharidoses (MPS). However, the incidence and severity of OSA with respect to disease type is poorly defined. The aim of the present study was to measure objectively the degree of OSA in a group of children with a range of MPS syndromes. Methods: In a cross-sectional study, cardiopulmonary sleep studies were performed during unsedated sleep in 26 children with MPS over a period of 2 years. Scores of OSA severity based upon clinical history and upon objective sleep study data were made in each case and compared. Results: OSA was present in 24/26 patients, and ranged in severity from mild to severe. OSA was most marked in MPS type IH (Hurler syndrome) followed by types IHS (Hurler–Scheie syndrome) and II (Hunter syndrome). Frequent arousals and poor sleep quality, not suspected clinically, were noted in several patients. There was agreement between the clinical and objective scoring systems in only 17/26 patients (65%) with clinical history scores tending to underestimate the most severe cases (5/26 cases) and overestimate the severity in the mild cases (4/26 cases). Conclusions: Obstructive respiratory problems are frequent in MPS patients and there are differences in severity of OSA between the different MPS types. Assessments of the severity of OSA based upon clinical history alone are inadequate. Our results suggest that objective sleep studies are necessary to evaluate these cases, to monitor clinical outcome and to assess the effects of therapeutic intervention. Prospective studies in larger numbers of patients are needed to validate these observations.
Identifier
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<a href="http://doi.org/10.1016/S0165-5876(01)00417-7" target="_blank" rel="noreferrer noopener">10.1016/S0165-5876(01)00417-7</a>
Rights
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Article information provided for research and reference use only. PedPalASCNET does not hold any rights over the resource listed here. All rights are retained by the journal listed under publisher and/or the creator(s).
2001
breathing difficulties
characteristics
Dinwiddie R
frequent awakenings
Galactosialidosis
International Journal of Pediatric Otorhinolaryngology
Lane R
Leighton SEJ
MPSI
MPSII
MPSIII
MPSIIIA
MPSIIIB
MPSVI
MPSVII
obstructive sleep apnea
OSA
Papsin B
poor sleep quality
sleep disturbance
Trajectory
Vellodi A