Characterization of swallowing biomechanics and function in untreated infants with spinal muscular atrophy: A natural history dataset
Rattachement africain : us, nz, br. Niveau de preuve : code pays fourni par la source.
Le résumé fourni par la source
BACKGROUND: Spinal Muscular Atrophy (SMA) is a progressive neuromuscular disorder that in its most severe form, causes profound swallowing deficits. There remains a paucity of research systematically elucidating the biomechanical and functional correlates. This void limits the ability to evaluate the effects of disease-modifying treatments on swallowing. OBJECTIVE: Elucidate characteristics of swallowing biomechanics and function among untreated patients with SMA. METHODS: Infants with SMA who had not received disease modifying therapy when they underwent a videofluoroscopic swallow study (VFSS) were retrospectively identified from 13 international children's hospitals. Infants were eligible if they exhibited symptoms by six months old, or in cases where they detected prior to symptom onset, if they had two copies of SMN2. Eligible infants underwent medical record review to gather oral intake status and need for suctioning for secretion management. Digital files of VFSS' were extracted and analyzed using BabyVFSSImP™. Non-parametric t-tests were used to compare swallowing biomechanics based on viscosity and the presence of dysphagia symptoms. RESULTS: 62 infants were included in this investigation with average age at initial VFSS 4.4 ± 3.11 months. A high proportion of infants exhibited profound deficits in swallowing biomechanics that impeded extraction of the bolus from the nipple, clearance of the bolus from the pharynx, and prevention of the bolus from entering the airway. Deficits were significantly more prevalent in infants referred for VFSS due to dysphagia symptoms than asymptomatic infants referred as part of high-risk referral (t ≤ 2.5, p ≤ 0.03). A high proportion of infants relied on suctioning for secretion management (34%; N = 21) and alternative nutrition (39%; N = 24), with those relying on these supports exhibiting significantly worse swallowing biomechanics than those who did not (t ≤ 2.8, p ≤ 0.01). CONCLUSIONS: Profound deficits in swallowing biomechanics and function are common in infants with SMA. Though clinical symptoms may aid in identifying infants with the most profound swallowing deficits, their absence in cases of more mild deficits inhibits the ability to use these clinical markers as a valid metric of swallowing integrity.
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Le contrôle bibliographique ouvert
DOI retrouvé dans Crossref DOI retrouvé ; titre concordant.
- Titre Crossref
- Characterization of swallowing biomechanics and function in untreated infants with spinal muscular atrophy: A natural history dataset
- Date Crossref
- 01/01/2025
- Éditeur
- SAGE Publications
- Type
- journal-article
Ce recoupement confirme des métadonnées liées au DOI. Il ne confirme ni la méthode ni les conclusions de l’étude, et il ne compte pas comme une seconde source scientifique indépendante.
Où se fait cette recherche
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University of Minnesota Department of Speech Language and Hearing Science pays non établi dans la noticeUniversité ou école supérieure
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University of Minnesota Children's Hospital pays non établi dans la noticeÉtablissement de santé
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Boston University pays non établi dans la noticeUniversité ou école supérieure
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Boston Children's Hospital Department of Critical Care Medicine pays non établi dans la noticeÉtablissement de santé
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University of Auckland Department of Speech Science pays non établi dans la noticeUniversité ou école supérieure
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Lurie Children's Hospital Department of Speech Language Pathology pays non établi dans la noticeÉtablissement de santé
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Nationwide Children's Hospital pays non établi dans la noticeÉtablissement de santé
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University of Florida Department of Neurology pays non établi dans la noticeUniversité ou école supérieure
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Florida College pays non établi dans la noticeUniversité ou école supérieure
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Primary Children's Hospital Department of Speech Language Pathology pays non établi dans la noticeÉtablissement de santé
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Children's Hospital of Philadelphia Department of Neurology pays non établi dans la noticeOrganisme public
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Universidade Federal do Rio Grande do Sul Department of Health and Human Communication pays non établi dans la noticeUniversité ou école supérieure
Department of Speech Language and Hearing Science — University of Minnesota, University of Minnesota Children's Hospital et Boston University, avec 9 autres affiliations.
Une affiliation ne permet pas de déduire la nationalité d’un auteur.