Swallowing markers in spinal and bulbar muscular atrophy

Banno, Haruhiko; Katsuno, Masahisa; Suzuki, Keisuke; Tanaka, Seiya; Suga, Noriaki; Hashizume, Akira; Mano, Tomoo; Araki, Atsuko; Watanabe, Haruo; Fujimoto, Yasushi; Yamamoto, Masahiko; Sobue, Gen

doi:10.1002/acn3.425

Cited by 14 publications

(13 citation statements)

References 37 publications

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“…Findings from fluoroscopic investigations examining the physiological correlates for these symptoms support these clinical reports of reduced severity of deficits. Videofluoroscopic results indicate patients with SMA Types II–IV typically have much lower rates of bolus airway entry ( Banno et al, 2017 ; Cha et al, 2010 ; Hashizume et al, 2017 ; van den Engel-Hoek et al, 2009 ). Though a case report of a 25-year-old patient with SMA Type II revealed impaired laryngeal closure resulting in aspiration ( Cha et al, 2010 ), other larger investigations have indicated none of their participants exhibited aspiration and 0%–26% of patients exhibited penetration ( Banno et al, 2017 ; van den Engel-Hoek et al, 2009 ).…”

Section: Review Of Pertinent Literaturementioning

confidence: 99%

“…Videofluoroscopic results indicate patients with SMA Types II–IV typically have much lower rates of bolus airway entry ( Banno et al, 2017 ; Cha et al, 2010 ; Hashizume et al, 2017 ; van den Engel-Hoek et al, 2009 ). Though a case report of a 25-year-old patient with SMA Type II revealed impaired laryngeal closure resulting in aspiration ( Cha et al, 2010 ), other larger investigations have indicated none of their participants exhibited aspiration and 0%–26% of patients exhibited penetration ( Banno et al, 2017 ; van den Engel-Hoek et al, 2009 ). Interpretation of these findings requires careful consideration to natural variants in deficits by SMA type, as well as findings from healthy, nondysphagic adults, which indicate 2% demonstrate aspiration and 9% demonstrate penetration on a videofluoroscopic swallow study ( Garand et al, 2019 ).…”

Section: Review Of Pertinent Literaturementioning

confidence: 99%

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Dysphagia Phenotypes in Spinal Muscular Atrophy: The Past, Present, and Promise for the Future

McGrattan

Graham

DiDonato

et al. 2021

Am J Speech Lang Pathol

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Purpose The aim of this study was to provide clinicians with an overview of literature relating to dysphagia in spinal muscular atrophy (SMA) to guide assessment and treatment. Method In this clinical focus article, we review literature published in Scopus and PubMed between 1990 and 2020 pertaining to dysphagia in SMA across the life span. Original research articles that were published in English were included. Searches were conducted within four themes of inquiry: (a) etiology and phenotypes, (b) respiratory systemic deficits and management, (c) characteristics of natural history dysphagia and its treatment, and (d) dysphagia outcomes with disease-modifying therapies. Articles for the first two themes were selected by content experts who identified the most salient articles that would provide clinicians foundational background knowledge about SMA. Articles for the third theme were identified using search terms, including spinal muscular atrophy, swallow, dysphagia, bulbar, nutrition, g-tube, alternative nutrition, jaw, mouth, palate, OR mandible . Search terms for the fourth theme included spinal muscular atrophy AND nusinersen OR AVXS-101/onasemnogene abeparvovec-xioi . Review of Pertinent Literature Twenty-nine articles were identified. Findings across identified articles support the fact that patients with SMA who do not receive disease-modifying therapy exhibit clinically significant deficits in oropharyngeal swallow function. Few investigations provided systematic information regarding the underlying physiological deficits responsible for this loss in function, the timing of the degradation, or how disease-modifying therapies change these outcomes. Conclusion Future research outlining the physiological and functional oropharyngeal swallowing deficits among patients with SMA who receive disease-modifying therapy is critical in developing standards of dysphagia care to guide clinicians.

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Section: Review Of Pertinent Literaturementioning

confidence: 99%

Section: Review Of Pertinent Literaturementioning

confidence: 99%

Dysphagia Phenotypes in Spinal Muscular Atrophy: The Past, Present, and Promise for the Future

McGrattan

Graham

DiDonato

et al. 2021

Am J Speech Lang Pathol

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“…Patients with spinal and bulbar muscular atrophy (SBMA), another type of motor neuron disease, also develop dysphagia in both oral and pharyngeal phases. In patients with SBMA, pharyngeal phase abnormalities are more frequently observed than oral phase abnormalities (Banno et al., ). Patients with Parkinson’s disease, one of the most common neurodegenerative diseases, also frequently develop dysphagia.…”

Section: Discussionmentioning

confidence: 99%

Oral phase dysphagia in facial onset sensory and motor neuronopathy

et al. 2018

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IntroductionFacial onset motor and sensory neuronopathy (FOSMN) is a rare disease whose cardinal features are initial asymmetrical facial sensory deficits followed by bulbar symptoms and spreading of sensory and motor deficits from face to scalp, neck, upper trunk, and upper extremities in a rostral–caudal direction. Although bulbar involvement is frequently observed in FOSMN, dysphagia in these patients has not been fully described. In this study, we aimed to characterize dysphagia as a prognostic factor in FOSMN by investigating our institutional case series.MethodsWe retrospectively reviewed the medical records, including swallowing function tests, of six patients with FOSMN (three men and three women) who were thoroughly examined at Kyushu University Hospital between 1 January 2005 and 30 November 2017.ResultsAverage age at onset was 58.5 years; average disease duration was 5.7 years. All patients developed bulbar dysfunction and dysphagia (at an average of 1.8 and 2.6 years from onset, respectively), resulting in choking episodes in three patients, percutaneous endoscopic gastrostomy placement in three, and recurrent aspiration pneumonia in one. Four of five patients evaluated with videofluoroscopic swallowing studies had poor oral retention, leading to bolus flowing into the pharynx before swallowing; the fifth patient showed poor lingual transfer. Fiberoptic endoscopic evaluation of swallowing revealed leakage of blue‐dyed water from the mouth to the pharynx in three patients because of poor oral retention, but only mild pharyngeal phase dysphagia in all four cases evaluated.ConclusionsOral phase dysphagia predominates in the early stage of FOSMN.

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“…Early diagnosis of dysphagia in PD may prevent hospitalization, disability, deaths, and global burden 13 for which periodical evaluation of oromotor function is required. Clinically early assessment is crucial for patient prognosis 14 and available gold standard tools videofluoroscopy (VFS) or Flexible endoscopic evaluations of swallowing (FEES) are still with constraints 2,15,16,17,18,19 . If such diagnosis is complemented with non-invasive, patient and environment friendly objective investigation, it will facilitate early identification, optimal intervention with better prognosis and reducing the morbidity.…”

Section: Introductionmentioning

confidence: 99%

Subtle oromotor signs in early Parkinson’s disease before clinical manifestations of dysphagia

Gudwani

Upadhyay

Sharma

et al. 2021

Preprint

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Background: Swallowing is complex phenomena comprising oral (preparatory and pre-swallow positioning), oropharyngeal, pharyngeal and esophageal phases. The timing of these phases is controlled by brain stem pattern generators including reflex for oropharyngeal propulsion and transit. Dysphagia in Parkinson's disease (PD) commonly observed at late stages with aspiration, pneumonia and hospitalization. Objective: Can subtle oromotor signs (if any) be observed for planning early interventions in PD Methods: The present study investigated oromotor function in fourteen early PD (onset ≤2years; H&Y ≤2.5) with dynamic MRI using saline (water) bolus and compared with seven age-matched healthy controls. Results: All the patients with PD were non-symptomatic for dysphagia by self-reporting, and on clinical assessment (Part-II MDS-UPDRS, Swallowing disturbance questionnaire, SDQ and Clinical assessment of dysphagia in neurodegeneration, CADN). Qualitatively MR images visualized, differences in PD compared to healthy controls for tongue-wave, velar-closure or release, bolus placement, oropharyngeal reflex-initiation, transit-time, epiglottic-closure-coordination and post-swallow oral or pharyngeal residue. Descriptive analysis showed higher variability of velar-closure, oropharyngeal- and pharygoesophageal-transit time in patient with PD. Group analysis (two-sample) show significant difference for velar-closure. Conclusion: Multiple lingual-waves, reverse-tongue thrust, with delayed velar control attributed to incoordinated muscular rhythm. Variable oropharyngeal transit time (0.64 to 2.25 msec) in PD ascribed to brainstem degenerative changes. Findings imply that subtle observable early oromotor signs as pre-clinical manifestation when evaluated with non-invasive, non-contrast dynamic MRI support early intervention, to prevent late-stage aspiration episodes and consequent hospitalizations.

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Swallowing markers in spinal and bulbar muscular atrophy

Cited by 14 publications

References 37 publications

Dysphagia Phenotypes in Spinal Muscular Atrophy: The Past, Present, and Promise for the Future

Dysphagia Phenotypes in Spinal Muscular Atrophy: The Past, Present, and Promise for the Future

Oral phase dysphagia in facial onset sensory and motor neuronopathy

Subtle oromotor signs in early Parkinson’s disease before clinical manifestations of dysphagia

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