High Resolution Manofluorographic Study in Patients With Multiple System Atrophy: Possible Early Detection of Upper Esophageal Sphincter and Proximal Esophageal Abnormality

Ueha, Rumi; Goto, Takao; Sato, Taku; Nativ‐Zeltzer, Nogah; Shen, S. C.; Nito, Takaharu; Belafsky, Peter C.; Yamasoba, Tatsuya

doi:10.3389/fmed.2018.00286

Cited by 15 publications

(23 citation statements)

References 19 publications

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“…In cases of pharyngeal dysphagia, impaired UES relaxation often occurs in Parkinson's disease and MSA (3, 7). Inadequate duration of UES relaxation and abnormal hyper-pressure of the resting UES are often recognized in patients with MSA regardless of the MSA type (4, 7). Impaired laryngeal elevation and reduced pharyngeal contraction can often be observed in patients with ALS resulting from weakness of the pharyngeal muscles with disease progression (5).…”

Section: Discussionmentioning

confidence: 99%

“…The videofluoroscopic swallowing study (VFSS) showed no apparent impairment in the oral phase of swallowing, although mild dysarthria was observed. High-resolution manofluorography (HRMF) showed deficient relaxation of the UES during swallowing and abnormal deglutitive proximal esophageal contraction (Figure 1B) (4). To secure an air way and maintain it clear, aspiration prevention surgery was considered.…”

Section: Case Reportsmentioning

confidence: 99%

“…Dysphagia is an important complication in patients with neuromuscular disorders, including Parkinson's disease and amyotrophic lateral sclerosis (ALS); it negatively impacts patient quality of life and may lead to fatal outcome due to malnutrition and aspiration pneumonia (1, 2). It is reported that there are various underlying mechanisms of dysphagia in neuromuscular diseases: deficient relaxation of the upper esophageal sphincter (UES) during swallowing in patients with Parkinson's disease (3), abnormal deglutitive proximal esophageal contraction and insufficient UES opening in patients with multiple system atrophy (MSA) (4), and weakness of pharyngeal contraction and impaired laryngeal elevation in patients with ALS (5). It has been reported that aspiration prevention surgery for severe dysphagia can improve the quality of life of patients and their families and help patients regain oral intake (6–8).…”

Section: Introductionmentioning

confidence: 99%

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Choice of Aspiration Prevention Surgery for Patients With Neuromuscular Disorders: Report of Three Cases

et al. 2019

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Dysphagia, one of the major complications of neuromuscular diseases such as Parkinson's disease and amyotrophic lateral sclerosis (ALS), decreases quality of life and may lead to malnutrition or aspiration pneumonia. Although recent reports have suggested that surgical aspiration prevention improves quality of life and enables oral intake, the selection of appropriate aspiration prevention techniques has rarely been discussed. In this report, we present the cases of three patients with neuromuscular diseases who underwent surgical aspiration prevention; we selected the surgical techniques based on analysis of the dysphagia mechanisms, disease progression, and general condition in each case. Case 1 was a 55-year-old man with multiple system atrophy (MSA) and presented with dysphagia associated with insufficient upper esophageal sphincter (UES) relaxation. We performed central-part laryngectomy, which was able to improve UES relaxation. Case 2 was a 79-year-old man with progressive supranuclear palsy who presented with respiratory disorder and dysphagia. Glottic closure under local anesthesia was selected because he also had acute hepatobiliary dysfunction and methicillin-resistant Staphylococcus aureus pneumonia with pleural effusion. Case 3 was a 75-year-old man with ALS and presented with respiratory disorder and mild dysphagia. Subglottic closure with total cricoidectomy was selected because his dysphagia was expected to worsen due to tracheostomy and disease progression. We also summarize the characteristics of the aspiration prevention surgical techniques based on our cases and on literature review. The causes of dysphagia, including insufficient UES opening during swallowing, weak pharyngeal constriction, velopharyngeal insufficiency, and inadequate laryngeal elevation, should be assessed by detailed examination before surgery, and the type of aspiration prevention surgery should be selected based on patient swallowing function and general condition.

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Section: Discussionmentioning

confidence: 99%

Section: Case Reportsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Choice of Aspiration Prevention Surgery for Patients With Neuromuscular Disorders: Report of Three Cases

et al. 2019

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“…Although the oropharyngeal phases of swallowing are most impaired in MSA, subtle esophageal dysmotility may also occur. Main dysfunctions observed by means of VFSS and high-resolution manometry, even in patients who were asymptomatic, included food stagnation in the esophagus, reduced esophageal peristalsis, hypomotility of the distal esophagus, abnormally high upper esophageal sphincter (UES) pressure and uncoordinated proximal esophageal contraction pressure during swallowing and resting [23,27,28].…”

Section: Diagnosis Of Dysphagia In Msamentioning

confidence: 99%

Dysphagia in multiple system atrophy consensus statement on diagnosis, prognosis and treatment

Calandra‐Buonaura

Alfonsi

Vignatelli

et al. 2021

Parkinsonism & Related Disorders

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Multiple system atrophy (MSA) is a neurodegenerative disorder characterized by a combination of autonomic failure plus cerebellar syndrome and/or parkinsonism. Dysphagia is a frequent and disabling symptom in MSA and its occurrence within 5 years of motor onset is an additional diagnostic feature. Dysphagia can lead to aspiration pneumonia, a recognized cause of death in MSA. Guidelines for diagnosis and management of dysphagia in MSA are lacking. An International Consensus Conference among experts with methodological support was convened in Bologna to reach consensus statements for the diagnosis, prognosis, and treatment of dysphagia in MSA. Abnormalities of the oral and pharyngeal phases of swallowing, esophageal dysfunction and aspiration occur in MSA and worsen as the disease progresses. According to the consensus, dysphagia should be investigated through available screening questionnaires and clinical and instrumental assessment (videofluoroscopic study or fiberoptic endoscopic evaluation of swallowing and manometry) at the time of MSA diagnosis and periodically thereafter. There is evidence that dysphagia is associated with poor survival in MSA, however effective treatments for dysphagia are lacking. Compensatory strategies like diet modification, swallowing maneuvers and head postures should be applied and botulinum toxin injection may be effective in specific conditions. Percutaneous endoscopic gastrostomy may be performed when there is a severe risk of malnutrition and pulmonary complications, but its impact on survival is undetermined. Several research gaps and unmet needs for research involving diagnosis, prognosis, and treatment were identified.

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“…Previously ADPEC was defined as abnormal hypertensive and discoordinated abnormal proximal esophageal contraction during swallowing evaluated by HRMF 14 (Fig. 1).…”

Section: Abnormal Deglutitive Proximal Esophageal Contractionmentioning

confidence: 99%

Esophageal Dysmotility is Common in Patients With Multiple System Atrophy

et al. 2020

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Objectives: Esophageal dysmotility (ED) in patients with multiple system atrophy (MSA) are poorly understood. This study aimed to investigate the prevalence of ED in patients with MSA and to assess the relationship of esophageal abnormalities with other clinical findings and characteristics in these patients. Methods: A retrospective chart review was conducted to identify patients with MSA and to compare them to the elderly controls without MSA (65+ years) who underwent a videofluorographic esophagram from 2014 to 2019. Disease type, disease severity, vocal fold mobility impairment, abnormal deglutitive proximal esophageal contraction (ADPEC), and intra-esophageal stasis (IES) were reviewed and compared between groups. Results: Thirty-seven patients with MSA were identified. The median age was 63 and 26 (70%) were male. These patients were matched to 22 elderly adults with presbylarynx but not MSA (median age 77, 68% male). In MSA patients, cerebellar variant type was predominant (59%), and ADPEC was recognized in 18 patients (49%). Disease severity level (P = 0.028) and existence of IES (P = 0.046) were associated with higher risks of developing ADPEC. The prevalence of IES was significantly higher in patients with MSA (95%) compared to controls without MSA (46%) (P < 0.001). Disease severity level and the existence of IES were significantly associated with the presence of ADPEC (p < 0.05). Conclusion: ADPEC and IES were significantly more common in MSA than in elderly subjects without MSA. MSA severity is associated with the development of ADPEC. The data suggest that esophageal motility is predominantly affected in MSA.

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High Resolution Manofluorographic Study in Patients With Multiple System Atrophy: Possible Early Detection of Upper Esophageal Sphincter and Proximal Esophageal Abnormality

Cited by 15 publications

References 19 publications

Choice of Aspiration Prevention Surgery for Patients With Neuromuscular Disorders: Report of Three Cases

Choice of Aspiration Prevention Surgery for Patients With Neuromuscular Disorders: Report of Three Cases

Dysphagia in multiple system atrophy consensus statement on diagnosis, prognosis and treatment

Esophageal Dysmotility is Common in Patients With Multiple System Atrophy

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