Efficacy of LSVT LOUD® on Phonatory Control and Voice Quality in Patients with Primary Progressive Apraxia of Speech: Case Studies.

Primary progressive apraxia of speech (PPAOS) is a neurodegenerative syndrome characterized by the progressive and initially isolated or predominant onset of difficulties in the planning/programming of movements necessary for speech production and can be accompanied by dysarthria. To date, no study has used an evidence-based treatment to address phonation control in patients with PPAOS. The aim of this study was to evaluate the feasibility and efficacy of LSVT LOUD® as a treatment for phonatory control in speakers with PPAOS. Three speakers with PPAOS received LSVT LOUD® therapy, and changes in phonatory control, voice quality and prosody were measured immediately, and one, four and eight weeks after the end of the treatment. Overall, the results suggest that the treatment is feasible and could improve voice quality, intensity, and control in some patients with PPAOS. The generalization of the results is also discussed.

Telerehabilitation for Lee Silverman Voice Treatment (Tele-LSVT)-Loud on voice intensity and voice use in daily living in people with multiple sclerosis: A protocol for a feasibility and pilot randomized controlled study.

Objective: Alterations in voice intensity and quality may constitute a social life limitation in people with multiple sclerosis (MS), but only 2% of cases receive speech therapy. Especially the Lee Silverman Voice Treatment (LSVT)-Loud is a highly effective intensive method for voice intensity, requiring subjects' repeated attendance at the clinic. Telerehabilitation may represent a feasible solution to bypass potential barriers related to speech therapy attendance, scaling up the beneficial effects of the treatment to a broader population. The proposed protocol aims to test the feasibility and the pilot efficacy of the LSVT-Loud delivered in telerehabilitation (Tele-LSVT-Loud), compared to the same treatment delivered in the clinic (LSVT-Loud). Methods: A single-blinded, parallel, two-arm, pilot randomized (1:1 ratio) controlled trial will be performed involving 20 people with MS. Patients will be allocated to 4 weeks of Tele-LSVT-Loud by accessing a telerehabilitation platform at home or LSVT-Loud conventionally delivered in the clinic. Feasibility and pilot effectiveness will be evaluated three times: before (T0), after the treatment (T1), and 3-month follow-up (T2). Feasibility measures will include adherence, adverse events, user experience, motivation, engagement, and acceptability. Vocal intensity during a 1-minute monologue will be the primary outcome measure. Secondary outcome measures will be the vocal quality during a 1-minute monologue, sustained /a/ voice intensity, quality and stability, voice use in daily life, voice subjective perception in daily life, and quality of life. Results: Expected results will be (1) high feasibility of Tele-LSVT-Loud and (2) a non-inferiority effect of Tele-LSVT-Loud compared with face-to-face treatment delivery on voice intensity and quality outcomes. Conclusions: Tele-LSVT-Loud may be a feasible intervention for MS alteration in voice intensity and quality with a non-inferior effect compared to LSVT-Loud.

Premotor cortex is hypoactive during sustained vowel production in individuals with Parkinson's disease and hypophonia.

Introduction: Hypophonia is a common feature of Parkinson's disease (PD); however, the contribution of motor cortical activity to reduced phonatory scaling in PD is still not clear. Methods: In this study, we employed a sustained vowel production task during functional magnetic resonance imaging to compare brain activity between individuals with PD and hypophonia and an older healthy control (OHC) group. Results: When comparing vowel production versus rest, the PD group showed fewer regions with significant BOLD activity compared to OHCs. Within the motor cortices, both OHC and PD groups showed bilateral activation of the laryngeal/phonatory area (LPA) of the primary motor cortex as well as activation of the supplementary motor area. The OHC group also recruited additional activity in the bilateral trunk motor area and right dorsal premotor cortex (PMd). A voxel-wise comparison of PD and HC groups showed that activity in right PMd was significantly lower in the PD group compared to OHC (p < 0.001, uncorrected). Right PMd activity was positively correlated with maximum phonation time in the PD group and negatively correlated with perceptual severity ratings of loudness and pitch. Discussion: Our findings suggest that hypoactivation of PMd may be associated with abnormal phonatory control in PD.

Self-perceived affective, behavioral, and cognitive reactions associated with voice use in people with Parkinson's disease: a pilot study.

PURPOSE: This study aimed to compare the affective, behavioral, and cognitive reactions related to vocal function in people with Parkinson's disease (PWPD) and healthy controls using the Behavior Assessment Battery - Voice (BAB-Voice). The test's internal consistency was also described. METHODS: 31 PWPD and 19 healthy controls were recruited from September 2020 to March 2021. Participants completed four BAB-Voice subtests: Speech Situation Checklist - Emotional Reaction (SSC-ER), the Speech Situation Checklist - Speech Disruption (SSC-SD), Behavior Checklist (BCL), and Communication Attitude Test for Adults (BigCAT), describing the experienced negative emotional reaction, voice disruptions, coping behaviors, and negative attitude regarding communication respectively. Subtest scores were calculated and analyzed. RESULTS: The scores of the PWPD were significantly different from those of the controls (Pillai's Trace = 0.344, F[4] = 5.508, p = .001, ηp2 = .344): PWPD showed more negative emotions and voice problems, more coping behaviors, and more negative speech-related attitude compared to healthy controls. All subtests showed excellent internal consistency. CONCLUSIONS: The BAB-Voice proved a tool with a good internal consistency that measured different psychosocial reactions in PWPD versus controls. PWPD exhibited significantly more negative emotions and voice problems in specific speech situations, more coping behaviors, and a more negative speech-related attitude. The specificity of information obtained from the BAB-Voice may aid in improving the treatment planning of voice disorders in PWPD.

Conversational Vocal Intensity in Parkinson's Disease: Treatment and Environmental Comparisons.

BACKGROUND: Vibrotactile Feedback (VF) using wearable devices is an emerging treatment option for hypophonia in Individuals with Parkinson's disease (IwPD). Studies evaluating the effectiveness of VF in improving conversational vocal intensity in real-life environment in IwPD are limited. OBJECTIVE: To determine the effect of VF on conversational vocal intensity and compare vocal intensity between a) clinic and real-life environment b) VF and Lee Silverman Voice Treatment (LSVT LOUD®)vs. VF alone in IwPD using a portable voice monitor (VocaLog2). METHODS: Eight individuals with hypophonia secondary to PD were randomly assigned to two treatment groups- VF and LSVT LOUD® (Group 1) and VF (Group 2). VF was provided using VocaLog2 device. Duration of treatment was 4 weeks for both groups. Vocal intensity was measured in the real-life environment at baseline, during treatment, and at one-month follow-up. Vocal intensity in clinic was obtained at baseline and one-month follow-up. Voice Handicap Index (VHI) questionnaire was administered at baseline and one-month follow-up. RESULTS: There was no significant difference in conversational vocal intensity between a) clinic and real-life environment at any point of time b) baseline and follow up for both treatment groups c) the two treatment groups at baseline, during each of the 4 weeks of treatment and at follow up d) VHI baseline and one month follow up scores. CONCLUSION: VF, including when combined with LSVT LOUD®, is limited in improving conversational vocal intensity in real-life in IwPD. The effects of frequency and duration of VF on conversational vocal intensity must be systematically investigated using large scale studies in IwPD.

Partner perception of affective, behavioral, and cognitive reactions to voice use in people with Parkinson's disease.

Introduction: People with Parkinson's disease (PWPD) experience negative feelings, thoughts, and coping behaviors due to the experienced communication challenges. This study aimed to compare the perceptions of PWPD with those of proxies for the affective, behavioral, and cognitive reactions specific to voice production during communicative interactions. Methods: The Behavior Assessment Battery - Voice (BAB-Voice) was administered to 31 PWPD and their close communication partner/proxy. The BAB-Voice contained four subtests: Speech Situation Checklist - Emotional Reaction (SSC-ER), Speech Situation Checklist - Speech Disruption (SSC-SD), Behavior Checklist (BCL), and Communication Attitude Test for Adults (BigCAT). The scores for each of these subtests were calculated and statistically analyzed. Results: A repeated measures MANOVA did not find statistically significant differences between the subscores of PWPD and proxies (Pillai's trace = 0.25, F[4] = 2.22, p =.094, ηp 2  = 0.25). Fair to excellent agreement between the PWPD and proxies was found. The highest agreement was found on the BigCAT (ICC = 0.80). The SSC-SD (ICC = 0.77) and SSC-ER (ICC = 0.71) still showed excellent agreement, while only fair agreement was found for the BCL (ICC = 0.57). Conclusion: Proxies were able to identify the affective, behavioral, and cognitive reactions to voice use in PWPD. Communication partners close to the PWPD could, therefore, provide valuable information regarding the assessment and treatment of hypophonia in PD.

The effects of a simulated fMRI environment on voice intensity in individuals with Parkinson's disease hypophonia and older healthy adults.

PURPOSE: Functional magnetic resonance imaging (fMRI) has promise for understanding neural mechanisms of neurogenic speech and voice disorders. However, performing vocal tasks within the fMRI environment may not always be analogous to performance outside of the scanner. Using a mock MRI scanner, this study examines the effects of a simulated scanning environment on vowel intensity in individuals with Parkinson's disease (PD) and hypophonia and older healthy control (OHC) participants. METHOD: Thirty participants (15 PD, 15 OHC) performed a sustained /ɑ/ vowel production task in three conditions: 1) Upright, 2) Mock Scanner + No Noise, and 3) Mock Scanner + MRI noise. We used a linear mixed-effects (multi-level) model to evaluate the contributions of group and recording environment to vowel intensity. A second linear mixed-effects model was also used to evaluate the contributions of PD medication state (On vs. Off) to voice intensity. RESULTS: Vowel intensity was significantly lower for PD compared to the OHC group. The intensity of vowels produced in the Upright condition was significantly lower compared to the Mock Scanner + No Noise condition, while vowel intensity in the Mock Scanner + MRI Noise condition was significantly higher compared to the Mock Scanner + No Noise condition. A group by condition interaction also indicated that the addition of scanner noise had a greater impact on the PD group. A second analysis conducted within the PD group showed no effects of medication state on vowel intensity. CONCLUSION: Our findings demonstrate that performance on voice production tasks is altered for PD and OHC groups when translated into the fMRI environment, even in the absence of acoustic scanner noise. For fMRI studies of voice in PD hypophonia, careful thought should be given to how the presence of acoustic noise may differentially affect PD and OHC, for both group and task comparisons.

Deficits in monitoring self-produced speech in Parkinson's disease.

OBJECTIVE: Speech deficits are common in Parkinson's disease, and behavioural findings suggest that the deficits may be due to impaired monitoring of self-produced speech. The neural mechanisms of speech deficits are not well understood. We examined a well-documented electrophysiological correlate of speech self-monitoring in patients with Parkinson's disease and control participants. METHODS: We measured evoked electroencephalographic responses to self-produced and passively heard sounds (/a/ phonemes) in age-matched controls (N = 18), and Parkinson's disease patients who had minor speech impairment, but reported subjectively experiencing no speech deficits (N = 17). RESULTS: During speaking, auditory evoked activity 100 ms after phonation (N1 wave) was less suppressed in Parkinson's disease than controls when compared to the activity evoked by passively heard phonemes. This difference between the groups was driven by increased amplitudes to self-produced phonemes, and reduced amplitudes passively heard phonemes in Parkinson's disease. CONCLUSIONS: The finding indicates that auditory evoked activity is abnormally modulated during speech in Parkinson's patients who do not subjectively notice speech impairment. This mechanism could play a role in producing speech deficits in as the disease progresses. SIGNIFICANCE: Our study is the first to show abnormal early auditory electrophysiological correlates of monitoring speech in Parkinson's disease patients.

Efficacy and Acceptance of a Lombard-response Device for Hypophonia in Parkinson's Disease.

OBJECTIVE: The purpose of this study was to examine the effectiveness, satisfaction, and acceptance of a low-cost Lombard-response (LR) device in a group of individuals with Parkinson's disease (IWPD) and their communication partners (CPs). METHOD: Sixteen IWPD and hypophonia and their CPs participated in the study. The IWPD wore a LR device that included a small MP3 player (Sony Walkman) and headphones playing a multi-talker noise audio file at 80 dB during lab-based speech tasks and during their daily conversational speech over a 2-week device trial period. Outcome measures included average conversational speech intensity and scores on a questionnaire related to speech impairment, communication effectiveness, and device satisfaction. RESULTS: Conversational speech intensity of the IWPD is increased by 7 to 10 dB with the LR device. Following a 2-week trial period, eight of the IWPD (50%) gave the LR device moderate-to-high satisfaction and effectiveness ratings and decided to purchase the device for long-term daily use. At the 4-month follow-up, none of the IWPDs were still using the LR device. Device rejection was related to discomfort (loudness), headaches, interference with cognition, and difficulty controlling device. CONCLUSION: Short-term acceptance and satisfaction with the LR device was moderate, but long-term acceptance, beyond 4 months, was absent. Future studies are required to determine if other types of low-cost LR devices can be developed that improve long-term efficacy and device acceptance in IWPD and hypophonia.

Auditory Dysfunction in Parkinson's Disease.

PD is a progressive and complex neurological disorder with heterogeneous symptomatology. PD is characterized by classical motor features of parkinsonism and nonmotor symptoms and involves extensive regions of the nervous system, various neurotransmitters, and protein aggregates. Extensive evidence supports auditory dysfunction as an additional nonmotor feature of PD. Studies indicate a broad range of auditory impairments in PD, from the peripheral hearing system to the auditory brainstem and cortical areas. For instance, research demonstrates a higher occurrence of hearing loss in early-onset PD and evidence of abnormal auditory evoked potentials, event-related potentials, and habituation to novel stimuli. Electrophysiological data, such as auditory P3a, also is suggested as a sensitive measure of illness duration and severity. Improvement in auditory responses following dopaminergic therapies also indicates the presence of similar neurotransmitters (i.e., glutamate and dopamine) in the auditory system and basal ganglia. Nonetheless, hearing impairments in PD have received little attention in clinical practice so far. This review summarizes evidence of peripheral and central auditory impairments in PD and provides conclusions and directions for future empirical and clinical research. © 2020 International Parkinson and Movement Disorder Society.

Subthalamic deep brain stimulation aggravates speech problems in Parkinson's disease: Objective and subjective analysis of the influence of stimulation frequency and electrode contact location.

BACKGROUND: Speech disorders, including stuttering and hypophonia, have been reported in patients with Parkinson's disease (PD) after subthalamic deep brain stimulation (STN-DBS). OBJECTIVE: To evaluate the effect of stimulation frequency or electrode contact location on speech disorders in PD patients with STN-DBS. METHOD: In this case-controlled study, we enrolled 50 PD patients with, and 100 PD patients without STN-DBS to compare their vocal intensities, measured by a sound pressure meter, and perceptual speech ratings, obtained from the speech sections of the United Parkinson's Disease Rating Scale (UPDRS) and subjective ratings regarding the impediment of functional communication by stuttering. For patients with STN-DBS, comparisons were made between high-frequency (HFS; 130 Hz), low-frequency (LFS; 80 Hz), and off-stimulation. We also evaluated the effect of electrode contact locations on speech function. RESULTS: Patients with STN-DBS had decreased vocal intensities and UPDRS scores compared to those without (p < 0.05). Vocal intensity was significantly lower during HFS than during LFS and off-stimulation (both, p < 0.05). Stuttering impeded STN-DBS patients' communication to greater extent than for those without (p < 0.001). Vocal intensity was lower when active contacts were in the dorsal zone compared to those in the ventral zone (p < 0.05). Only STN-DBS treatment was a predictive factor for low vocal intensity (OR = 9.53, p = 0.04). CONCLUSION: High-frequency STN-DBS with dorsal zone contacts can aggravate certain speech problems in PD patients. Therefore, it is important to balance between motor control and speech impairments in these patients.

Hypophonia as only presenting symptom in myasthenia gravis - a diagnostic dilemma in poor countries: a case report.

INTRODUCTION: The autoimmune disease myasthenia gravis can mimic a variety of neurological disorders leading to a delay in diagnosis and treatment. In the older population, due to confusion with signs of the ageing process or comorbidities due to ageing, there are many underdiagnosed or misdiagnosed cases of myasthenia gravis. A majority of myasthenia gravis symptoms appear as ocular or motor symptoms and there are very few cases of bulbar symptoms. We present a case of myasthenia gravis with only hypophonia as a clinical feature. CASE PRESENTATION: We present a case of a 51-year-old Madheshi woman whose only complaint was sudden onset of hypophonia which later showed a fluctuating nature throughout the daytime. There was only reduced pitch in her voice with no nasal tone or dysarthria (so-called dysphonia), which created a diagnostic dilemma. Later, a neurological examination and other relevant investigations suggested myasthenia gravis. CONCLUSIONS: Sudden onset and focal neurological deficit can raise the diagnostic dilemma of myasthenia gravis. Relevant laboratory tests and clinical examinations are important to diagnose this disease properly. In resources-deprived nations like Nepal, where several investigations are expensive and access to them is difficult, it becomes very difficult to achieve a solid diagnosis for rare presentations of diseases.

Speech disorders in Parkinson's disease: pathophysiology, medical management and surgical approaches.

The prevalence of speech disorders among individuals with Parkinson's disease (PD) has been reported to be as high as 89%. Speech impairment in PD results from a combination of motor and nonmotor deficits. The production of speech depends upon the coordination of various motor activities: respiration, phonation, articulation, resonance and prosody. A speech disorder is defined as impairment in any of its inter-related components. Despite the high prevalence of speech disorders in PD, only 3-4% receive speech treatment. Treatment modalities include pharmacological intervention, speech therapy, surgery, deep brain stimulation and vocal fold augmentation. Although management of Parkinsonian dysarthria is clinically challenging, speech treatment in PD should be part of a multidisciplinary approach to patient care in this disease.

Hypophonia as a sign of thalamus lesion: a case report.

Hypophonia is a neurological sign usually seen after brainstem or peripheral damage, either at the recurrent laryngeal nerve or vocal cord level. However, it has been described as a sign of supratentorial strokes in a few studies, specifically when anterior and ventral thalamic involvement is reported. In addition, it is a prominent sign of other neurological disorders such as Parkinson disease and other extrapyramidal conditions. We describe a case of hypophonia secondary to a left thalamic hemorrhage, after a careful search of other potential causes of this deficit, and we discuss the underlying neuroanatomical circuits.

Neuroanatomical Correlates of Hypophonia in Subcortical Vascular Cognitive Impairment.

BACKGROUND/AIMS: Early detection and intervention may alter the disease course of subcortical vascular cognitive impairment (SVCI). Patients with SVCI have white matter ischemia that disrupts connections between the cortex and subcortical gray matter and therefore manifest various symptoms such as motor disturbances and behavioral/cognitive dysfunction. Reduced vocal loudness, or hypophonia, is one of the common motor symptoms of SVCI, but few studies have systematically investigated it in this patient population. The main purpose of this investigation was to identify neural pathways underlying hypophonia in patients with SVCI. METHODS: Eighty-eight patients with SVCI and 21 normal controls performed phonation tasks. Diffusion tensor imaging data from 73 patients were utilized to measure white matter changes associated with hypophonia. RESULTS: Correlational analyses between white matter fractional anisotropy values and the decibel level of the "sustained phonation" task identified the left midbrain cerebral peduncle (corticobulbar tract), external capsule, corona radiata/internal capsule, and bilateral frontal white matter as possible neural correlates for hypophonia. CONCLUSION: Our results support the notion that hypophonia in SVCI patients might be caused by the impairment of the pyramidal and extrapyramidal systems. This study provides a unique contribution towards understanding the neuropathology of hypophonic features in this population.

Reduction of Parkinson's-related dysphonia by thyroplasty.

Parkinson's-related dysphonia has a negative impact on the quality of speech by increasing the effects of the associated dysarthria. When this dysphonia is related to vocal fold adduction defect, constituting a real glottic insufficiency, vocal fold medialization can be proposed after failure of intensive voice and speech therapy. Acoustic and aerodynamic voice and speech analysis techniques, perceptual evaluation and estimation of vocal handicap, associated with fiberoptic laryngoscopy were performed to determine the indication for vocal fold medialization in these patients with glottic insufficiency. Vocal fold medialization by Montgomery thyroplasty implant was performed under local anesthesia and neuroanalgesia in two patients with Parkinson's disease presenting a dysphonia refractory to speech therapy. Postoperative evaluation showed improvement of voice quality with an increased number of harmonics and improvement of aerodynamic parameters. Vocal fold medialization by Montgomery thyroplasty implant effectively improved voice quality in these two patients allowing a more effective vocal fold adduction. The reducing of the hypophonia has a positive effect on the quality of oral communication. The medialization thyroplasty technique, under local anesthesia, allows intraoperative control of the voice as well as removal of the implant when necessary.

[Neurorehabilitation in Parkinson's disease]. / Neurorehabilitation bei der Parkinson-Krankheit.

Parkinson's disease (PD) is a chronic neurodegenerative disease which is characterized by the cardinal symptoms akinesia, rigidity, rest tremor and postural instability. Besides PD features also a wide range of non-motor symptoms. Physical activity is recommended for all stages of PD and may hypothetically even have a positive influence on the course of the disease. Rehabilitative treatments become increasingly important in the advanced stage of the disease and include mainly physiotherapy, occupational therapy and speech therapy. Neurorehabilitation is arguably most important for the treatment of axial symptoms such as freezing, hypophonia, dysphagia, postural instability and postural disturbances that respond poorly to drug therapy. This article provides an overview of current developments in the field of neurorehabilitation in PD.

Examining the relationship between speech intensity and self-rated communicative effectiveness in individuals with Parkinson's disease and hypophonia.

PURPOSE: To examine the relationship between speech intensity and self-ratings of communicative effectiveness in speakers with Parkinson's disease (PD) and hypophonia. An additional purpose was to evaluate if self-ratings of communicative effectiveness made by participants with PD differed from ratings made by primary communication partners. METHODS: Thirty participants with PD and 15 healthy older adults completed the Communication Effectiveness Survey. Thirty primary communication partners rated the communicative effectiveness of his/her partner with PD. Speech intensity was calculated for participants with PD and control participants based on conversational utterances. RESULTS: Results revealed significant differences between groups in conversational speech intensity (p=.001). Participants with PD self-rated communicative effectiveness significantly lower than control participants (p=.000). Correlational analyses revealed a small but non-significant relationship between speech intensity and communicative effectiveness for participants with PD (r=0.298, p=.110) and control participants (r=0.327, p=.234). Self-ratings of communicative effectiveness made participants with PD was not significantly different than ratings made by primary communication partners (p=.20). CONCLUSIONS: Obtaining information on communicative effectiveness may help to broaden outcome measurement and may aid in the provision of educational strategies. Findings also suggest that communicative effectiveness may be a separate and a distinct construct that cannot necessarily be predicted from the severity of hypophonia.

Pathomechanisms and compensatory efforts related to Parkinsonian speech.

Voice and speech in Parkinson's disease (PD) patients are classically affected by a hypophonia, dysprosody, and dysarthria. The underlying pathomechanisms of these disabling symptoms are not well understood. To identify functional anomalies related to pathophysiology and compensation we compared speech-related brain activity and effective connectivity in early PD patients who did not yet develop voice or speech symptoms and matched controls. During fMRI 20 PD patients ON and OFF levodopa and 20 control participants read 75 sentences covertly, overtly with neutral, or with happy intonation. A cue-target reading paradigm allowed for dissociating task preparation from execution. We found pathologically reduced striato-prefrontal preparatory effective connectivity in early PD patients associated with subcortical (OFF state) or cortical (ON state) compensatory networks. While speaking, PD patients showed signs of diminished monitoring of external auditory feedback. During generation of affective prosody, a reduced functional coupling between the ventral and dorsal striatum was observed. Our results suggest three pathomechanisms affecting speech in PD: While diminished energization on the basis of striato-prefrontal hypo-connectivity together with dysfunctional self-monitoring mechanisms could underlie hypophonia, dysarthria may result from fading speech motor representations given that they are not sufficiently well updated by external auditory feedback. A pathological interplay between the limbic and sensorimotor striatum could interfere with affective modulation of speech routines, which affects emotional prosody generation. However, early PD patients show compensatory mechanisms that could help improve future speech therapies.