Behavioral Management of Respiratory/Phonatory Dysfunction for Dysarthria Associated With Neurodegenerative Disease: A Systematic Review.

PURPOSE: This systematic review represents an update to previous reviews of the literature addressing behavioral management of respiratory/phonatory dysfunction in individuals with dysarthria due to neurodegenerative disease. METHOD: Multiple electronic database searches and hand searches of prominent speech-language pathology journals were conducted in accordance with Preferred Reporting Items for Systematic Reviews and Meta-Analyses standards. RESULTS: The search yielded 1,525 articles, from which 88 met inclusion criteria and were reviewed by two blinded co-investigators. A large range of therapeutic approaches have been added to the evidence base since the last review, including expiratory muscle strength training, singing, and computer- and device-driven programs, as well as a variety of treatment modalities, including teletherapy. Evidence for treatment in several different population groups-including cerebellar ataxia, myotonic dystrophy, autosomal recessive spastic ataxia of Charlevoix-Saguenay, Huntington's disease, multiple system atrophy, and Lewy body dementia-were added to the current review. Synthesis of evidence quality provided strong evidence in support of only one behavioral intervention: Lee Silverman Voice Treatment Program (LSVT LOUD) in people with Parkinson's disease. No other treatment approach or population included in this review demonstrated more than limited evidence, reflecting that these approaches/populations require urgent further examination. CONCLUSION: Suggestions about where future research efforts could be significantly strengthened and how clinicians can apply research findings to their practice are provided. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.24964473.

Cough and Swallowing Therapy and Their Effects on Vocal Fold Bowing and Laryngeal Lesions.

OBJECTIVE: Expiratory muscle strength training (EMST) and sensorimotor training of airway protection (smTAP) are two exercises intended to improve cough and swallowing in people with Parkinson's Disease (PwPD). The aims of this study were to (1) examine whether EMST or smTAP elicit changes to vocal fold bowing; and (2) describe the safety of EMST and smTAP as it relates to the development of vocal fold lesions. METHOD(S): This was a secondary analysis of data from PwPD who completed EMST or smTAP as part of a prospective randomized controlled trial. Vocal fold bowing (BI) and the presence of laryngeal lesions were blindly analyzed from flexible endoscopic evaluation of swallowing (FEES) using ImageJ software and operational definitions. Linear regression was used to examine the influence time (pre- vs. post-therapy) and therapy (EMST vs. smTAP) on vocal fold bowing. Descriptive statistics were used to describe the presence of laryngeal lesions. RESULT(S): Overall, 56 participants were included, 28 per group. The median BI scores pre- and post-therapy were 8.2% and 8.3% for the EMST group and 11.3% and 8.4% for the smTAP group, respectively. Statistical analyses revealed insufficient evidence to suggest an effect of time and treatment type on BI (p > 0.05) or on the presence of vocal fold lesions (p > 0.05). CONCLUSION: Based on these and previous findings, it appears that changes in vocal fold bowing do not drive treatment effects following EMST and smTAP. Also, this study further supports the safety of smTAP and EMST despite the required forceful exhalation and repetitive coughing. LEVEL OF EVIDENCE: 4 Laryngoscope, 134:1127-1132, 2024.

Auditory-Perceptual Assessments of Cough: Characterizing Rater Reliability and the Effects of a Standardized Training Protocol.

INTRODUCTION: Auditory-perceptual assessments of cough are commonly used by speech-language pathologists working with people with swallowing disorders with emerging evidence beginning to demonstrate their validity; however, their reliability among novice clinicians is unknown. Therefore, the primary aim of this study was to characterize the reliability of auditory-perceptual assessments of cough among a group of novice clinicians. As a secondary aim, we assessed the effects of a standardized training protocol on the reliability of auditory-perceptual assessments of cough. METHODS: Twelve novice clinicians blindly rated ten auditory-perceptual cough descriptors for 120 cough audio clips. Standardized training was then completed by the group of clinicians. The same cough audio clips were then re-randomized and blindly rated. Reliability was analyzed pre- and post-training within each clinician (intra-rater), between each unique pair of raters (dyad-level inter-rater), and for the entire group of raters (group-level inter-rater) using intraclass correlation coefficients and Cohen's Kappa. RESULTS: Pre-training reliability was greatest for measures of strength, effectiveness, and normality and lowest when judging the type of expiratory maneuver (cough, throat clear, huff, other). The measures that improved the most with training were ratings of perceived crispness, amount of voicing, and type of expiratory maneuver. Intra-rater reliability coefficients ranged from 0.580 to 0.903 pre-training and 0.756-0.904 post-training. Dyad-level inter-rater reliability coefficients ranged from 0.295 to 0.745 pre-training and 0.450-0.804 post-training. Group-level inter-rater reliability coefficients ranged from 0.454 to 0.919 pre-training and 0.558-0.948 post-training. CONCLUSION: Reliability of auditory-perceptual assessments varied across perceptual cough descriptors, but all appeared within the range of what has been historically reported for auditory-perceptual assessments of voice and visual-perceptual assessments of swallowing and cough airflow. Reliability improved for most cough descriptors following 30-60 min of standardized training. Future research is needed to examine the validity of auditory-perceptual assessments of cough by assessing the relationship between perceptual cough descriptors and instrumental measures of cough effectiveness to better understand the role of perceptual assessments in clinical practice.

Sensorimotor Cough Dysfunction in Cerebellar Ataxias.

Cerebellar ataxias are neurological conditions with a high prevalence of aspiration pneumonia and dysphagia. Recent research shows that sensorimotor cough dysfunction is associated with airway invasion and dysphagia in other neurological conditions and may increase the risk of pneumonia. Therefore, this study aimed to characterize sensorimotor cough function and its relationship with ataxia severity. Thirty-seven participants with cerebellar ataxia completed voluntary and/or reflex cough testing. Ataxia severity was assessed using the Scale for the Assessment and Rating of Ataxia (SARA). Linear multilevel models revealed voluntary cough peak expiratory flow rate (PEFR) estimates of 2.61 L/s and cough expired volume (CEV) estimates of 0.52 L. Reflex PEFR (1.82 L/s) and CEV (0.34 L) estimates were lower than voluntary PEFR and CEV estimates. Variability was higher for reflex PEFR (15.74% coefficient of variation [CoV]) than voluntary PEFR (12.13% CoV). 46% of participants generated at least two, two-cough responses following presentations of reflex cough stimuli. There was a small inverse relationship between ataxia severity and voluntary PEFR (ß = -0.05, 95% CI: -0.09 - -0.01 L) and ataxia severity and voluntary CEV (ß = -0.01, 95% CI: -0.02 - -0.004 L/s). Relationships between reflex cough motor outcomes (PEFR ß = 0.03, 95% CI: -0.007-0.07 L/s; CEV ß = 0.007, 95% CI: -0.004-0.02 L) and ataxia severity were not statistically robust. Results indicate that voluntary and reflex cough sensorimotor dysfunction is present in cerebellar ataxias and that increased severity of ataxia symptoms may impact voluntary cough function.

Normative Reference Values for FEES and VASES: Preliminary Data From 39 Nondysphagic, Community-Dwelling Adults.

PURPOSE: The aim of this study was to establish preliminary reference values for the Visual Analysis of Swallowing Efficiency and Safety (VASES)-a standardized rating methodology used to evaluate swallowing safety and efficiency for flexible endoscopic evaluation of swallowing (FEES). METHOD: FEES were completed in nondysphagic, community-dwelling adults using a standardized protocol of 15 swallowing trials that varied by bolus size, consistency, contrast agent, and swallowing instructions. FEES were blindly analyzed using VASES. Primary outcome measures included bolus location at swallow onset, Penetration-Aspiration Scale (PAS) scores, and percentage-based residue ratings for six anatomic landmarks. Secondary outcome measures included sip size, bite size, and number of swallows. RESULTS: Thirty-nine healthy adults completed the study, yielding an analysis of 584 swallows. Swallows were initiated with the bolus in the pharynx for 41.8% of trials. PAS 1 was the most common score, accounting for 75.3% of trials, followed by PAS 3, which accounted for 18.8% of trials. When residue was present (> 0%), the amount was relatively small across all anatomic landmarks, with median residue ratings of 2.0% (oropharynx), 1.5% (hypopharynx), 3.0% (epiglottis), 3.0% (laryngeal vestibule), and 3.5% (vocal folds). Five events of aspiration were observed, which were characterized by subglottic residue ratings of 1%, 3%, 10%, 24%, and 90%. The average sip size of self-selected volume cup sips of water was 19.8 ml, and the average bite size of a 3.0-g saltine cracker was 1.33 g. Moreover, 78% of the trials in this study protocol (except 90-ml trials) were completed in a single swallow. DISCUSSION: The results from this study provide preliminary norms for VASES that could be used as a reference when assessing functional swallowing outcomes during FEES. While this is an important first step in establishing norms for FEES and VASES, clinicians and researchers should be mindful that the normative reference values from this study are from a relatively small study sample (N = 39), with most people below the age of 60 years (n = 30). Future research should expand on these norms by including a greater number of people across the age continuum and with greater racial, ethnic, and gender diversity. Supplemental Material and Open Science Form: https://doi.org/10.23641/asha.23504325.

The Role of Aspiration Amount on Airway Protective Responses in People with Neurogenic Dysphagia.

The aim of this study was to examine relationships between the presence vs. absence of an aspiration-related airway protective response (i.e., coughing or throat clearing) with aspiration amount, trial volume, disease diagnosis, and disease duration in people with neurologic disease. A secondary analysis was completed of flexible endoscopic evaluations of swallowing (FEES) in people with neurologic disease. Thin liquid boluses with endoscopically confirmed aspiration were included. Aspiration amount was measured for each trial using the visual analysis of swallowing efficiency and safety (VASES). Statistical analyses were used to (1) compare aspiration amount between swallows with vs. without an airway protective response and (2) examine if trial volume, disease duration, and disease diagnosis were related to the presence of airway protective responses when controlling for aspiration amount. 422 aspirated swallows across 86 FEES were analyzed. Of the 59 people who aspirated more than once, 66.1% exhibited variability in the presence vs. absence of an airway protective response. Statistical analyses revealed airway protective responses were significantly related to aspiration amount (p < 0.001; Marginal R2 = 0.46) and disease duration (p = 0.036, L.R. = 4.35) but not trial volume (p = 0.428) or disease diagnosis (p = 0.103). The participants in this study were less likely to cough or throat clear when having smaller amounts of aspiration or longer disease durations. Future research is needed to examine if aspiration amount is related to airway protective responses in healthy adults and across other patient populations.

Rehabilitating Cough Dysfunction in Parkinson's Disease: A Randomized Controlled Trial.

BACKGROUND: Disorders of airway protection (cough and swallowing) are pervasive in Parkinson's disease (PD) resulting in a high incidence of aspiration pneumonia and death. However, there are no randomized controlled trials comparing strength and skill-based approaches to improve airway protection in PD. OBJECTIVES: The aim of this study was to compare expiratory muscle strength training (EMST) and sensorimotor training for airway protection (smTAP) to improve cough-related outcomes in people with PD. METHODS: Participants with PD and dysphagia were recruited for this prospective phase II randomized-blinded controlled clinical trial. Participants completed baseline assessment, 5 weeks of EMST or smTAP, and a post-training assessment. Primary outcome measures included maximum expiratory pressure (MEP) and voluntary cough peak expiratory flow rate (PEFR). Mixed effects models were used to assess the effects of EMST and smTAP on outcomes. RESULTS: A total of 65 participants received either EMST (n = 34) or smTAP (n = 31). MEP improved from pre- to post-treatment for smTAP (P < 0.001, d = 0.19) and EMST (P < 0.001, d = 0.53). Voluntary PEFR increased from pre- to post-treatment for smTAP (P < 0.001, d = 0.19) and EMST (P < 0.001, d = 0.06). Moreover, reflex cough PEFR (P < 0.001, d = 0.64), reflex cough expired volume (P < 0.001, d = 0.74), and urge to cough (P = 0.018, OR = 2.70) improved for the smTAP group but not for the EMST group. CONCLUSIONS: This clinical trial confirmed the efficacy of smTAP to improve reflex and voluntary cough function, above and beyond EMST, the current gold standard. © 2022 International Parkinson and Movement Disorder Society.

The Relationship Between Lingual Strength and Functional Swallowing Outcomes in Parkinson's Disease.

The purpose of this retrospective study was to determine whether reduced lingual strength was associated with functional swallowing outcomes in individuals with Parkinson's disease (PD). Participants (N = 42) completed evaluations of maximal lingual isometric pressure (MIP) and mean lingual swallowing pressure (MSP), and flexible endoscopic evaluations of swallowing. Regression models were used to determine the association between lingual strength and functional swallowing outcomes of airway invasion, the presence of post-swallow pharyngeal residue, and the amount of pharyngeal residue (when present). Results revealed that higher MIP (p = 0.002, OR 0.93) and higher MSP (p = 0.001 OR 0.88) were associated with less airway invasion of thin liquids. Both MIP and MSP were able to differentiate between those with and without dysphagia (MIP: AUC 0.7935, p = 0.001; MSP: AUC 0.75, p = 0.026). Neither MIP nor MSP was related to the presence of residue. However, when thin liquid oropharyngeal residue was present, both MIP (p < 0.001, OR 0.99) and MSP (p < 0.001; OR 0.98) were significantly associated with the amount of residue observed. Similarly, when thin liquid hypopharyngeal residue was present, MIP (p < 0.001, OR 0.99) and MSP (p < 0.001, OR 0.98) were associated with the amount of residue observed. These findings suggest a relationship between reduced lingual strength and worse thin liquid swallowing safety and efficiency; however, the magnitude of these effects was small. This indicates that lingual strength is one important contributing factor to functional swallowing impairments in PD and may identify those with unsafe swallowing. These findings have important clinical implications for including lingual strength in the screening, assessment, and management of dysphagia in PD.

Rehabilitation of Airway Protection in Individuals With Movement Disorders: A Telehealth Feasibility Study.

PURPOSE: Airway protective deficits (swallowing and cough) greatly reduce health and quality of life and are a pervasive consequence of neurodegenerative movement disorders. Expiratory muscle strength training (EMST) and cough skill training (CST) are two treatment approaches to improve airway protection; however, many patients are unable to access these treatments. Telehealth may improve access to care, but it remains unknown whether these treatments are feasible and efficacious via telehealth. This study aimed to determine the practical feasibility and preliminary treatment effect of EMST and CST via telehealth. METHOD: Twenty participants with movement disorders completed 4 weeks of EMST and 2 weeks of CST, including two clinician-directed treatment sessions via telehealth and 3 days of home practice per week. Feasibility was calculated for each treatment. Practical feasibility was defined as completing treatment (EMST or CST) and obtaining the relevant outcome measures-a proxy of maximum expiratory pressure (pMEP) for EMST and peak expiratory flow rate (PEFR) for CST-within a 30-min session/period. Session factors that may have influenced feasibility were examined. Preliminary treatment effect was defined as changes in pMEP and PEFR. RESULTS: Time taken to obtain pMEP and complete EMST was 17.48 min, and time taken to obtain PEFR and complete CST was 17.69 min. pMEP, single voluntary cough PEFR, and sequential voluntary cough PEFR increased from pre- to posttreatment. CONCLUSIONS: Findings suggest that the delivery of EMST and CST is feasible via telehealth and yield improvements to pMEP and PEFR. This has important implications for expanding service delivery of airway protective interventions and reducing health care disparities in people with neurodegenerative movement disorders. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.21357669.

Visual Analysis of Swallowing Efficiency and Safety (VASES): A Standardized Approach to Rating Pharyngeal Residue, Penetration, and Aspiration During FEES.

The primary aim of this study was to describe the development of the Visual Analysis of Swallowing Efficiency and Safety (VASES)-a standardized method to rate pharyngeal residue, penetration, and aspiration during FEES. As a secondary aim, we explored the feasibility of training novices to interpret FEES using VASES. Literature review and consensus panel discussions were used to develop standardized rules for VASES. A training protocol was developed and criterion ratings were established. Twenty-five novice raters completed VASES training and pre-/post-training assessments. Statistical analyses were used to examine pre- to post-training differences in the accuracy, reliability, and time to rate each video clip using VASES. Four sets of VASES rules were developed, including 'what', 'where', 'when', and 'how' to rate FEES. Large, significant post-training improvements in rating accuracy were observed across all seven VASES outcome measures (Cohen's d range 0.74-1.59). Additionally, inter-rater reliability increased for four of the seven outcome measures, and the amount of time to rate each video clip decreased from 2.6 min pre-training to 1.5 min post-training. VASES is a standardized FEES rating method used to enhance the subjective analysis of pharyngeal residue, penetration, and aspiration. It can be feasibly taught to novice raters with a high level of success and may be an effective method to analyze swallowing safety and efficiency in clinical and research practices. Future research is needed to test the validity of VASES by examining its relationship with other validated FEES rating scales.

Characterizing Quality of Life in Caregivers of People with Parkinson's Disease and Dysphagia.

Caring for a family member with dysphagia can negatively impact caregiver wellbeing, although little is known about how dysphagia severity or specific symptoms influence this. The purpose of this study was to examine how objective measures of dysphagia in people with Parkinson's disease influenced their caregivers' quality of life. Fifty caregivers (mainly spouses) of people with Parkinson's disease completed a caregiver quality of life survey. Results were compared to medical chart reviews, interviews, and instrumental evaluations of swallowing from the care recipients. Outcomes included caregiver quality of life score, ratings of airway invasion and pharyngeal residue, and Parkinson's disease duration. Descriptive and regression analyses were completed. All caregivers reported reduced quality of life, with 28% having severely disturbed adaptation. Every care recipient with Parkinson's disease demonstrated airway invasion and/or pharyngeal residue. Together, the combination of older care recipient age and longer disease duration was associated with poorer caregiver quality of life [adj. R2 = 0.10-0.12, p = 0.03-0.4]. Neither airway invasion nor pharyngeal residue was related to caregiver quality of life (p > 0.05). Findings confirmed that caregivers of people with Parkinson's disease and dysphagia experience reduced quality of life; however, current methods of assessing caregivers' quality of life may not adequately account for dysphagia-specific burden. Results highlight the urgent need for the development of dysphagia-specific assessments of caregivers' quality of life to facilitate identification of high-risk caregivers and aid the development of support systems to improve health outcomes for caregivers and care recipients.

Effects of Bolus Holding on Respiratory-Swallow Coordination in Parkinson's Disease.

PURPOSE: The aim of this study was to examine the effects of bolus holding on respiratory-swallow coordination (RSC) in people with Parkinson's disease (PD). METHOD: People with PD were prospectively recruited to undergo RSC assessment using simultaneous respiratory inductive plethysmography and flexible laryngoscopy. During RSC assessment, participants swallowed 5-ml thin liquid boluses during held and nonheld swallowing tasks. Measures of RSC were analyzed for each swallow, which included respiratory pause duration, lung volume at swallow initiation, respiratory phase patterning, and the presence of paradoxical respiratory movements. Multilevel statistical modeling was used to determine if differences in RSC were present between the held and nonheld tasks. RESULTS: Thirty-three participants were enrolled. When compared to the nonheld swallows, the held swallows exhibited shorter respiratory pauses (p = .001, R 2 = .019), lower lung volumes at swallow initiation (p < .001, R 2 = .116), more frequent exhale-swallow-exhale patterns (p < .001, OR = 4.30), and less frequent paradoxical respiratory movements (p = .001, OR = 0.43). CONCLUSIONS: Findings from this study revealed that bolus holding significantly influences RSC in people with PD. This demonstrates that bolus holding may be an efficacious strategy to immediately improve RSC in PD. However, clinicians and researchers should consider avoiding bolus holding during swallowing evaluations if attempting to assess RSC behaviors that are most typical for the examinee.

Detection of Aspiration, Penetration, and Pharyngeal Residue During Flexible Endoscopic Evaluation of Swallowing (FEES): Comparing the Effects of Color, Coating, and Opacity.

The aim of this study was to assess the effects of color, coating, and opacity on the detection of aspiration, penetration, and residue during flexible endoscopic evaluations of swallowing (FEES). Thirty dysphagic adults underwent FEES while swallowing five 5 mL thin liquid boluses (1 × each, randomized): white water, blue water, white milk, blue milk, and barium water. To assess the effects of color, blue milk was compared to white milk. To assess the effects of coating, barium, white water, and white milk were compared to each other. To assess the effects of opacity, blue milk was compared to blue water. Videos were blindly analyzed and judged for the presence of pharyngeal residue, penetration, and aspiration. Repeated measures analyses were used to assess differences in the frequency of detection across bolus types. Pharyngeal residue was detected more frequently for liquids that were blue, had a coating effect, or were opaque (p < 0.05) when compared to liquids that were white, did not have a coating effect, or were translucent, respectively. Penetration and aspiration were detected more frequently for liquids that had a coating effect (p < 0.05), but not for liquids that were colored blue or opaque (p > 0.05). Coating appears to be the most important factor detecting thin liquid residue, penetration, and aspiration during FEES. Given these findings, standardized use of boluses that possess a coating effect (e.g., white-dyed water or barium) is highly recommended to enhance the sensitivity of identifying impairments in swallowing safety and efficiency during FEES.

Respiratory-Swallow Coordination Training and Voluntary Cough Skill Training: A Single-Subject Treatment Study in a Person With Parkinson's Disease.

Purpose Airway protective disorders are common in Parkinson's disease (PD), yet effective methods to rehabilitate these life-threatening impairments are limited. This study examined the effects of two skill-based treatments aimed at improving swallowing and cough in a severely dysphagic person with PD: respiratory-swallow coordination training (RSCT) and voluntary cough skill training (VCST). It was hypothesized that (a) RSCT would improve respiratory-swallow coordination and swallowing safety and efficiency and (b) VCST would improve reflex and voluntary cough effectiveness. Method An 81-year-old man with midstage PD and severe dysphagia was recruited for study participation. The study utilized a multiple-baseline ABACA experimental design with a 2-month delayed retention assessment. Measures of respiratory-swallow coordination, swallowing safety and efficiency, and cough effectiveness were collected at each assessment using respiratory inductive plethysmography, flexible endoscopic evaluations of swallowing, and spirometry. Data were analyzed descriptively using baseline corrected tau and standard mean difference effect sizes (d). Results Large effect sizes were observed immediately following RSCT for respiratory-swallow coordination (d = 9.17), penetration-aspiration (d = 12.88), vallecular residue (d = 1.75), piriform residue (d = 4.15), and overall dysphagia severity (d = 1.83). Large effect sizes were also observed immediately following VCST for single voluntary cough (d = 4.30), sequential voluntary cough (d = 3.28), and reflex cough (d = 5.58). Improvements were maintained 2 months later for all outcome measures except single voluntary cough. Discussion This is the first study to examine the effects of RSCT and VCST in a person with PD. Robust improvements in respiratory-swallow coordination and swallowing safety and efficiency were achieved following four sessions of RSCT, and significant improvements in reflex and voluntary cough strength were seen following four sessions of VCST. Future work is needed to study these treatments in larger cohorts of people with PD.