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Laryngoscope Investigative... Aug 2023This study aimed to systematically review the associations between motor clinical phenotypes in Parkinson's disease (PD) and laryngeal disease symptoms. Laryngeal... (Review)
Review
OBJECTIVE
This study aimed to systematically review the associations between motor clinical phenotypes in Parkinson's disease (PD) and laryngeal disease symptoms. Laryngeal dysfunctions such as dysphonia and dysphagia are ubiquitous in people with Parkinson's disease (PwPD). Similar to other disease symptoms, they manifest variably across PwPD. Some of the variability within PD has been explained by clinical phenotypes. However, it is unclear how laryngeal symptoms of PD express themselves across these phenotypes.
METHODS
Five databases were searched (MEDLINE, CINAHL, Web of Science, Embase, Scopus) in May 2022. After the removal of duplicates, all retrieved records were screened. Cohort, case-control, and cross-sectional studies in English discussing laryngeal symptoms and clinical PD phenotypes were included. Data were extracted, tabulated, and assessed using Moola et al.'s (2021) appraisal tool for systematic reviews of risk and etiology.
RESULTS
The search retrieved 2370 records, representing 540 PwPD. After the removal of duplicates and screening, eight articles were included for review. The most common phenotype categories were tremor-dominant and postural-instability gait disordered (PIGD). Five studies addressed vocal characteristics, while four considered swallowing. Differences and lack of rigor in methodology across studies complicated conclusions, but a tendency for tremor-dominant phenotypes to present with less severe laryngeal symptoms was found.
CONCLUSION
Some minor differences in laryngeal function were found between tremor-dominant and PIGD phenotypes in PD. However, there is a need for more standardized and high-quality studies when comparing motor phenotypes for laryngeal function.
PubMed: 37621279
DOI: 10.1002/lio2.1112 -
Annals of Clinical and Translational... Jan 2021X-linked Charcot-Marie-Tooth type 1 (CMTX1) is an inherited peripheral neuropathy caused by mutations in the gap junction beta 1 (GJB1) gene, which encodes the...
OBJECTIVE
X-linked Charcot-Marie-Tooth type 1 (CMTX1) is an inherited peripheral neuropathy caused by mutations in the gap junction beta 1 (GJB1) gene, which encodes the connexin32 protein. A small number of patients with GJB1 mutations present with episodic neurological dysfunction and reversible white matter lesions, which has not been adequately reported. Here, we aim to enable clinicians to further understand this particular situation through systematically reviewing all published relevant cases.
METHODS
We conducted a comprehensive search of the PubMed electronic database for medical literature relevant to CMTX1 patients with episodic neurological dysfunction and then fully analyzed the general information, clinical manifestations, and characteristics of magnetic resonance imaging (MRI), cerebrospinal fluid (CSF) analysis, and nerve conduction study (NCS).
RESULTS
We identified 47 cases of CMTX1 associated with episodic central nervous system (CNS) dysfunction from 38 publications. CMTX1 patients experienced episodic CNS deficits at a young age, ranging from infancy to 26 years, and 45 (95.7%) of them were male. The CNS symptoms manifested as facial, lingual, or limb weakness in 44 (93.6%), dysarthria or dysphagia in 39 (83.0%), facial or limb numbness in 15 (31.9%), and ataxia in 10 (21.3%) patients. The duration of episodic symptoms ranged from 3 minutes to 6 months. Thirty (63.8%) CMTX1 cases have reported obvious predisposing factors, among which the most common factors were infection or fever (27.7%), travel to high altitude (12.8%), and intensive exercise (8.5%). As for brain MRI, most abnormal signals were found in bilateral deep white matter (88.9%) and corpus callosum (80.0%). In addition, most of the NCS results were abnormal, including prolonged latency, reduced amplitude, and slowed conduction velocity. The motor nerve conduction velocity (MNCV) of median nerve was the most detectable and valuable, ranging from 25 to 45 m/s.
INTERPRETATION
We have reported the most comprehensive summary of the demographic and clinical profile from 47 CMTX1 patients with episodic CNS deficits and provided new insight into the phenotype spectrum of CMTX1. We hope that our study can help clinicians make early diagnosis and implement the best prevention and treatment strategies for CMTX1 patients with episodic CNS deficits.
Topics: Central Nervous System Diseases; Charcot-Marie-Tooth Disease; Female; Humans; Male
PubMed: 33314704
DOI: 10.1002/acn3.51271 -
Neurological Sciences : Official... May 2021The insular cortex serves a wide variety of functions in humans, ranging from sensory and affective processing to high-level cognition. Hence, insular dysfunction may... (Review)
Review
BACKGROUND AND PURPOSE
The insular cortex serves a wide variety of functions in humans, ranging from sensory and affective processing to high-level cognition. Hence, insular dysfunction may result in several different presentations. Ischemic strokes limited to the insular territory are rare and deserve a better characterization, to be quickly recognized and to receive the appropriate treatment (e.g. thrombolysis).
METHODS
We reviewed studies on patients with a first-ever acute stroke restricted to the insula. We searched in the Medline database the keywords "insular stroke" and "insular infarction", to identify previously published cases. Afterwards, the results were divided depending on the specific insular region affected by the stroke: anterior insular cortex (AIC), posterior insular cortex (PIC) or total insula cortex (TIC). Finally, a review of the clinical correlates associated with each region was performed.
RESULTS
We identified 25 reports including a total of 49 patients (59.7 ± 15.5 years, 48% male) from systematic review of the literature. The most common clinical phenotypes were motor and somatosensory deficits, dysarthria, aphasia and a vestibular-like syndrome. Atypical presentations were also common and included dysphagia, awareness deficits, gustatory disturbances, dysautonomia, neuropsychiatric or auditory disturbances and headache.
CONCLUSIONS
The clinical presentation of insular strokes is heterogeneous; however, an insular stroke should be suspected when vestibular-like, somatosensory, speech or language disturbances are combined in the same patient. Further studies are needed to improve our understanding of more atypical presentations.
Topics: Aphasia; Cerebral Cortex; Dysarthria; Female; Humans; Male; Speech; Stroke
PubMed: 33575921
DOI: 10.1007/s10072-021-05109-1 -
Otolaryngology--head and Neck Surgery :... Mar 2023Deep brain stimulation (DBS) has considerable efficacy for the motor dysfunction of idiopathic Parkinson's disease (PD) on patient quality of life. However, the benefit...
OBJECTIVE
Deep brain stimulation (DBS) has considerable efficacy for the motor dysfunction of idiopathic Parkinson's disease (PD) on patient quality of life. However, the benefit of DBS on voice and speech quality remains controversial. We carried out a systematic review to understand the influence of DBS on parkinsonian dysphonia and dysarthria.
DATA SOURCES
A PubMed/MEDLINE and Cochrane systematic review was carried out following Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) and Population, Intervention, Comparison, Outcome, Timing, and Setting (PICOTS) statements.
REVIEW METHODS
Three investigators screened studies published in the literature from inception to May 2022. The following data were retrieved: age, demographic, sex, disease duration, DBS duration, DBS location, speech, and voice quality measurements.
RESULTS
From the 180 studies identified, 44 publications met the inclusion criteria, accounting for 866 patients. Twenty-nine studies focused on voice/speech quality in subthalamic DBS patients, and 6 included patients with stimulation of pallidal, thalamic, and zona incerta regions. Most studies (4/6) reported a deterioration of the vocal parameters on subjective voice quality evaluation. For speech, the findings were more contrasted. There was an important heterogeneity between studies regarding the voice and speech quality outcomes used to evaluate the impact of DBS on voice/speech quality.
CONCLUSION
The impact of DBS on voice and speech quality significantly varies between studies. The stimulated anatomical region may have a significant role since the stimulation of the pallidal area was mainly associated with voice quality improvement, in contrast with other regions. Future controlled studies comparing all region stimulation are needed to get reliable findings.
LEVEL OF EVIDENCE
Level III: evidence from evidence summaries developed from systematic reviews.
Topics: Humans; Parkinson Disease; Speech; Deep Brain Stimulation; Quality of Life; Subthalamic Nucleus; Dysphonia
PubMed: 36040825
DOI: 10.1177/01945998221120189 -
Health Science Reports Nov 2022Facial palsy is a rare complication of the COVID-19 infection. Herein, we conducted a systematic review of all published cases of facial palsy post-COVID-19 infection in...
BACKGROUND AND AIMS
Facial palsy is a rare complication of the COVID-19 infection. Herein, we conducted a systematic review of all published cases of facial palsy post-COVID-19 infection in an attempt to educate the general population and medical practitioners regarding the likely occurrence of facial palsy in COVID-19 patients, its detection, effective treatment plan, and prognosis of the condition.
METHODS
We searched PubMed, Google Scholar, and Directory of Open Access Journals (DOAJ) from December 1, 2019 to September 21, 2021.
RESULTS
We included 49 studies bearing accounts of 75 cases who had facial palsy. The mean age of patients was 42.9 ± 19.59 years, with a male-to-female ratio of 8:7. The majority of the cases were reported from Brazil ( = 14), USA ( = 9), Turkey ( = 9), and Spain ( = 9). Noticeably, 30.14% of COVID-19 patients were diagnosed with Guillain-Barré syndrome. In total, 22.97% of patients complained of bilateral facial paralysis ( = 17), whereas ipsilateral paralysis was observed in 77.03% ( = 57). These were common complaints of Lagophthalmos, otalgia, facial drooping, dysarthria, and compromised forehead wrinkling. The treatment regimen mainly included the use of corticosteroids ( = 51) (69.86%), antivirals ( = 23) (31.51%), IVIG ( = 18) (24.66%), antibiotics ( = 13) (17.81%), antiretroviral ( = 9) (12.33%), and antimalarial ( = 8) (10.96%) medications. In all, 35.62% of patients ( = 26) adhered to a combination of antiviral and corticosteroid-based therapy. Positive treatment outcomes were observed in 83.58% ( = 56) of cases. In contrast, 10 patients (14.93%) showed nonsignificant recovery, out of which 3 (4.48%) died from the disease.
CONCLUSION
The association of facial palsy with COVID-19 is controversial and therefore requires further investigation and published work to confirm a causal relationship. However, physicians should not overlook the likelihood of facial palsy post-COVID-19 infection and treat it accordingly.
PubMed: 36320650
DOI: 10.1002/hsr2.887 -
American Journal of Speech-language... Mar 2024This systematic review represents an update to previous reviews of the literature addressing behavioral management of respiratory/phonatory dysfunction in individuals...
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.
Topics: Humans; Neurodegenerative Diseases; Dysarthria; Speech Therapy; Voice Training; Parkinson Disease
PubMed: 38232176
DOI: 10.1044/2023_AJSLP-23-00274 -
Frontiers in Human Neuroscience 2022Augmentative and alternative communication brain-computer interface (AAC-BCI) systems are intended to offer communication access to people with severe speech and...
UNLABELLED
Augmentative and alternative communication brain-computer interface (AAC-BCI) systems are intended to offer communication access to people with severe speech and physical impairment (SSPI) without requiring volitional movement. As the field moves toward clinical implementation of AAC-BCI systems, research involving participants with SSPI is essential. Research has demonstrated variability in AAC-BCI system performance across users, and mixed results for comparisons of performance for users with and without disabilities. The aims of this systematic review were to (1) describe study, system, and participant characteristics reported in BCI research, (2) summarize the communication task performance of participants with disabilities using AAC-BCI systems, and (3) explore any differences in performance for participants with and without disabilities. Electronic databases were searched in May, 2018, and March, 2021, identifying 6065 records, of which 73 met inclusion criteria. Non-experimental study designs were common and sample sizes were typically small, with approximately half of studies involving five or fewer participants with disabilities. There was considerable variability in participant characteristics, and in how those characteristics were reported. Over 60% of studies reported an average selection accuracy ≤70% for participants with disabilities in at least one tested condition. However, some studies excluded participants who did not reach a specific system performance criterion, and others did not state whether any participants were excluded based on performance. Twenty-nine studies included participants both with and without disabilities, but few reported statistical analyses comparing performance between the two groups. Results suggest that AAC-BCI systems show promise for supporting communication for people with SSPI, but they remain ineffective for some individuals. The lack of standards in reporting outcome measures makes it difficult to synthesize data across studies. Further research is needed to demonstrate efficacy of AAC-BCI systems for people who experience SSPI of varying etiologies and severity levels, and these individuals should be included in system design and testing. Consensus in terminology and consistent participant, protocol, and performance description will facilitate the exploration of user and system characteristics that positively or negatively affect AAC-BCI use, and support innovations that will make this technology more useful to a broader group of people.
CLINICAL TRIAL REGISTRATION
https://www.crd.york.ac.uk/prospero/display_record.php?ID=CRD42018095345, PROSPERO: CRD42018095345.
PubMed: 35966988
DOI: 10.3389/fnhum.2022.952380 -
European Journal of Neurology Oct 2020Approximately 89% of patients with Parkinson's disease (PD) suffer from dysarthria. Lee Silverman Voice Treatment (LSVT), a behavioral therapy, aims to improve speech... (Meta-Analysis)
Meta-Analysis
Approximately 89% of patients with Parkinson's disease (PD) suffer from dysarthria. Lee Silverman Voice Treatment (LSVT), a behavioral therapy, aims to improve speech and voice functions. The objective was to assess the effectiveness of LSVT compared with other/no speech interventions for dysarthria in patients with PD. Electronic databases, including PubMed, Embase and the Cochrane Library, were searched. The publication date of all included studies was before 6 March 2020. Only randomized controlled trials (RCTs) that evaluated the LSVT intervention compared with other/no speech intervention were considered. The data obtained from the included studies were described and the mean differences were calculated. Eight RCTs were included in this meta-analysis comparing LSVT with other/no speech interventions. In the comparison of LSVT versus no intervention, vocal intensity for sustained 'Ah' phonation, reading the 'Rainbow passage', monologue and describing a picture increased by 8.87, 4.34, 3.25 and 3.31 dB, respectively, after 1 month of therapy. Compared with the respiratory therapy group, the LSVT group also showed significant improvement in vocal intensity for sustained 'Ah' phonation, reading the 'Rainbow passage' and monologue immediately after treatment (13.39, 6.66 and 3.19 dB). Positive improvement still existed after 24 months. There was no difference in the therapeutic effect between face-to-face and online LSVT. The effectiveness of LSVT for dysarthria in patients with PD was verified in these trials. However, future RCTs with sufficient participants are essential to evaluate the effectiveness of LSVT for dysarthria.
Topics: Dysarthria; Humans; Parkinson Disease; Speech Therapy; Treatment Outcome
PubMed: 32539227
DOI: 10.1111/ene.14399 -
JMIR Rehabilitation and Assistive... Oct 2023Speech intelligibility and speech comprehension for dysarthric speech has attracted much attention recently. Dysarthria is characterized by irregularities in the speed,... (Review)
Review
BACKGROUND
Speech intelligibility and speech comprehension for dysarthric speech has attracted much attention recently. Dysarthria is characterized by irregularities in the speed, strength, pitch, breath control, range, steadiness, and accuracy of muscle movements required for articulatory aspects of speech production.
OBJECTIVE
This study examined the contributions made by other studies involved in dysarthric speech comprehension. We focused on the modes of meaning extraction used in generalizing speaker-listener underpinnings in light of semantic ontology extraction as a desired technique, applied method types, speech representations used, and databases sourced from.
METHODS
This study involved a systematic literature review using 7 electronic databases: Cochrane Database of Systematic Reviews, Web of Science Core Collection, Scopus, PubMed, ACM, IEEE Xplore, and Google Scholar. The main eligibility criterion was the extraction of meaning from dysarthric speech using natural language processing or understanding approaches to improve on dysarthric speech comprehension. In total, out of 834 search results, 30 studies that matched the eligibility requirements were acquired following screening by 2 independent reviewers, with a lack of consensus being resolved through joint discussion or consultation with a third party. In order to evaluate the studies' methodological quality, the risk of bias assessment was based on the Cochrane risk-of-bias tool version 2 (RoB2) with 23 of the studies (77%) registering low risk of bias and 7 studies (33%) raising some concern over the risk of bias. The overall quality assessment of the study was done using TRIPOD (Transparent Reporting of a Multivariable Prediction Model for Individual Prognosis or Diagnosis).
RESULTS
Following a review of 30 primary studies, this study revealed that the reviewed studies focused on natural language understanding or clinical approaches, with an increase in proposed solutions from 2020 onwards. Most studies relied on speaker-dependent speech features, while others used speech patterns, semantic knowledge, or hybrid approaches. The prevalent use of vector representation aligned with natural language understanding models, while Mel-frequency cepstral coefficient representation and no representation approaches were applied in neural networks. Hybrid representation studies aimed to reconstruct dysarthric speech or improve comprehension. Comprehensive databases, like TORGO and UA-Speech, were commonly used in combination with other curated databases, while primary data was preferred for specific or unique research objectives.
CONCLUSIONS
We found significant gaps in dysarthric speech comprehension characterized by the lack of inclusion of important listener or speech-independent features in the speech representations, mode of extraction, and data sources used. Further research is therefore proposed regarding the formulation of models that accommodate listener and speech-independent features through semantic ontologies that will be useful in the inclusion of key features of listener and speech-independent features for meaning extraction of dysarthric speech.
PubMed: 37889538
DOI: 10.2196/44489 -
Journal of Clinical Neuroscience :... Jun 2024Essential tremor is a neurological condition associated with movement disorder with more prevalence among adult group of population. The burden of essential tremor is... (Review)
Review
BACKGROUND
Essential tremor is a neurological condition associated with movement disorder with more prevalence among adult group of population. The burden of essential tremor is peaking globally but with the advancement in the area of functional neurosurgery such as stereotactic thalamotomy, the quality of life of such patients can be improved drastically.
METHODS
This systemic review was conducted in accordance to the guidance of preferred Reporting items for Systematic Review and Meta-Analysis(PRISMA). Databases of "PubMed", "Embase", "Web of Science", "Cinhal Plus", and "Scopus" from inception till 2023 was undertaken. A combination of keywords, Medical Subject Headings (MeSH), and search terms such as Search strategy for PubMed search was as follows: "stereotactic thalamotomy" AND "essential tremor".
RESULTS
This systematic review analyzed 9 studies with a total of 274 patients of essential tremor patients. Unilateral thalamotomy was carried out among 268 patients and bilateral thalamotomy in rest of the patients. Vim and Vom nucleus were the site of thalamotmy with ventral intermedius nucleus being the major one. Ten different types of clinical tremor rating scales were used to assess pre operative and post operative improvement in the tremor scales of the individual patients. Dysarthria and limb weakness was noted post operative complication in majority of the cases.
CONCLUSION
Our study revealed that stereotactic thalamotomy provided good functional outcome in patients of essential tremor who underwent unilateral thalamotomy compared to bilateral thalamotomy. The positive outcome outweighs the complications in such functional surgery.
PubMed: 38824802
DOI: 10.1016/j.jocn.2024.05.036