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1.
BMJ ; 386: e078341, 2024 Jul 10.
Artigo em Inglês | MEDLINE | ID: mdl-38986549

RESUMO

OBJECTIVES: To assess the clinical effectiveness of two speech and language therapy approaches versus no speech and language therapy for dysarthria in people with Parkinson's disease. DESIGN: Pragmatic, UK based, multicentre, three arm, parallel group, unblinded, randomised controlled trial. SETTING: The speech and language therapy interventions were delivered in outpatient or home settings between 26 September 2016 and 16 March 2020. PARTICIPANTS: 388 people with Parkinson's disease and dysarthria. INTERVENTIONS: Participants were randomly assigned to one of three groups (1:1:1): 130 to Lee Silverman voice treatment (LSVT LOUD), 129 to NHS speech and language therapy, and 129 to no speech and language therapy. LSVT LOUD consisted of four, face-to-face or remote, 50 min sessions each week delivered over four weeks. Home based practice activities were set for up to 5-10 mins daily on treatment days and 15 mins twice daily on non-treatment days. Dosage for the NHS speech and language therapy was determined by the local therapist in response to the participants' needs (estimated from prior research that NHS speech and language therapy participants would receive an average of one session per week over six to eight weeks). Local practices for NHS speech and language therapy were accepted, except for those within the LSVT LOUD protocol. Analyses were based on the intention to treat principle. MAIN OUTCOME MEASURES: The primary outcome was total score at three months of self-reported voice handicap index. RESULTS: People who received LSVT LOUD reported lower voice handicap index scores at three months after randomisation than those who did not receive speech and language therapy (-8.0 points (99% confidence interval -13.3 to -2.6); P<0.001). No evidence suggests a difference in voice handicap index scores between NHS speech and language therapy and no speech and language therapy (1.7 points (-3.8 to 7.1); P=0.43). Patients in the LSVT LOUD group also reported lower voice handicap index scores than did those randomised to NHS speech and language therapy (-9.6 points (-14.9 to -4.4); P<0.001). 93 adverse events (predominately vocal strain) were reported in the LSVT LOUD group, 46 in the NHS speech and language therapy group, and none in the no speech and language therapy group. No serious adverse events were recorded. CONCLUSIONS: LSVT LOUD was more effective at reducing the participant reported impact of voice problems than was no speech and language therapy and NHS speech and language therapy. NHS speech and language therapy showed no evidence of benefit compared with no speech and language therapy. TRIAL REGISTRATION: ISRCTN registry ISRCTN12421382.


Assuntos
Disartria , Terapia da Linguagem , Doença de Parkinson , Fonoterapia , Humanos , Doença de Parkinson/complicações , Disartria/etiologia , Disartria/terapia , Disartria/reabilitação , Masculino , Feminino , Fonoterapia/métodos , Idoso , Terapia da Linguagem/métodos , Reino Unido , Pessoa de Meia-Idade , Resultado do Tratamento , Treinamento da Voz , Medicina Estatal
2.
J Med Case Rep ; 18(1): 319, 2024 Jul 04.
Artigo em Inglês | MEDLINE | ID: mdl-38961428

RESUMO

BACKGROUND: Myasthenic crisis (MC) is a life-threatening complication of myasthenia gravis (MG), necessitating ventilation. Achieving a safe and timely diagnosis of myasthenic crisis with atypical, isolated presentation is a considerable challenge particularly in elderly patients, where myasthenia gravis can present with isolated dysarthria in rare instances, giving a clinical impression of lacunar stroke. CASE PRESENTATION: We present a compelling case of a 73-year-old Caucasian female presenting with abrupt onset of isolated dysarthria. Despite initial treatment for a presumed lacunar stroke, subsequent evaluations led to her diagnosis of a myasthenic crisis. Within 72 h of admission, the patient developed dysphagia and shortness of breath, requiring supplemental oxygen. The case highlights the sequential progression of events from the atypical presentation of isolated dysarthria and its course to the management of a myasthenic crisis. CONCLUSION: Our reported case focuses on the discussion of myasthenia that mimicked a lacunar stroke and was finally diagnosed at a critical time of medical crisis. This case highlights the imperative notion that isolated dysarthria in elderly individuals warrants vigilant monitoring for possible myasthenia gravis, given the low incidence of lacunar stroke presenting with only dysarthria.


Assuntos
Disartria , Miastenia Gravis , Acidente Vascular Cerebral Lacunar , Humanos , Miastenia Gravis/diagnóstico , Miastenia Gravis/complicações , Idoso , Disartria/etiologia , Feminino , Diagnóstico Diferencial , Acidente Vascular Cerebral Lacunar/diagnóstico , Acidente Vascular Cerebral Lacunar/complicações , Inibidores da Colinesterase/uso terapêutico , Transtornos de Deglutição/etiologia , Transtornos de Deglutição/diagnóstico , Dispneia/etiologia
4.
Mol Genet Metab ; 142(3): 108510, 2024 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-38843620

RESUMO

BACKGROUND: Information about dysarthria and dysphagia in mitochondrial diseases (MD) is scarce. However, this knowledge is needed to identify speech and swallowing problems early, to monitor the disease course, and to develop and offer optimal treatment and support. This study therefore aims to examine the prevalence and severity of dysarthria and dysphagia in patients with MD and its relation to clinical phenotype and disease severity. Secondary aim is to determine clinically relevant outcome measures for natural history studies and clinical trials. METHODS: This retrospective cross-sectional medical record study includes adults (age ≥ 18 years) diagnosed with genetically confirmed MD who participated in a multidisciplinary admission within the Radboud center for mitochondrial medicine between January 2015 and April 2023. Dysarthria and dysphagia were examined by administering the Radboud dysarthria assessment, swallowing speed, dysphagia limit, test of mastication and swallowing solids (TOMASS), and 6-min mastication test (6MMT). The disease severity was assessed using the Newcastle mitochondrial disease scale for adults (NMDAS). RESULTS: The study included 224 patients with MD with a median age of 42 years of whom 37.5% were male. The pooled prevalence of dysarthria was 33.8% and of dysphagia 35%. Patients with MD showed a negative deviation from the norm on swallowing speed, TOMASS (total time) and the 6MMT. Furthermore, a significant moderate relation was found between the presence of dysarthria and the clinical phenotypes. There was a statistically significant difference in total time on the TOMASS between the clinical phenotypes. Finally, disease severity showed a significant moderate relation with the severity of dysarthria and a significant weak relation with the severity of dysphagia. CONCLUSION: Dysarthria and dysphagia occur in about one-third of patients with MD. It is important for treating physicians to pay attention to this subject because of the influence of both disorders on social participation and wellbeing. Referral to a speech and language therapist should therefore be considered, especially in patients with a more severe clinical phenotype. The swallowing speed, TOMASS and 6MMT are the most clinically relevant tests to administer.


Assuntos
Transtornos de Deglutição , Disartria , Doenças Mitocondriais , Humanos , Transtornos de Deglutição/etiologia , Transtornos de Deglutição/fisiopatologia , Disartria/etiologia , Disartria/fisiopatologia , Masculino , Feminino , Doenças Mitocondriais/complicações , Doenças Mitocondriais/fisiopatologia , Adulto , Pessoa de Meia-Idade , Estudos Retrospectivos , Estudos Transversais , Idoso , Índice de Gravidade de Doença , Prevalência , Deglutição , Adulto Jovem , Fenótipo
5.
Am J Speech Lang Pathol ; 33(4): 1930-1951, 2024 Jul 03.
Artigo em Inglês | MEDLINE | ID: mdl-38838243

RESUMO

PURPOSE: This study investigated the effects of the SPEAK OUT! & LOUD Crowd therapy program on speaking rate, percent pause time, intelligibility, naturalness, and communicative participation in individuals with Parkinson's disease (PD). METHOD: Six adults with PD completed 12 individual SPEAK OUT! sessions across four consecutive weeks followed by group-based LOUD Crowd sessions for five consecutive weeks. Most therapy sessions were conducted via telehealth, with two participants completing the SPEAK OUT! portion in person. Speech samples were recorded at six time points: three baseline time points prior to SPEAK OUT!, two post-SPEAK OUT! time points, and one post-LOUD Crowd time point. Acoustic measures of speaking rate and percent pause time and listener ratings of speech intelligibility and naturalness were obtained for each time point. Participant self-ratings of communicative participation were also collected at pre- and posttreatment time points. RESULTS: Results showed significant improvement in communicative participation scores at a group level following completion of the SPEAK OUT! & LOUD Crowd treatment program. Two participants showed a significant decrease in speaking rate and increase in percent pause time following treatment. Changes in intelligibility and naturalness were not statistically significant. CONCLUSIONS: These findings provide preliminary support for the effectiveness of the SPEAK OUT! & LOUD Crowd treatment program in improving communicative participation for people with mild-to-moderate hypokinetic dysarthria secondary to PD. This study is also the first to demonstrate positive effects of this treatment program for people receiving the therapy via telehealth.


Assuntos
Doença de Parkinson , Inteligibilidade da Fala , Medida da Produção da Fala , Fonoterapia , Humanos , Doença de Parkinson/complicações , Doença de Parkinson/terapia , Masculino , Feminino , Idoso , Pessoa de Meia-Idade , Fonoterapia/métodos , Disartria/etiologia , Disartria/terapia , Disartria/reabilitação , Resultado do Tratamento , Acústica da Fala , Fatores de Tempo , Qualidade da Voz , Telemedicina
6.
Am J Speech Lang Pathol ; 33(4): 1952-1964, 2024 Jul 03.
Artigo em Inglês | MEDLINE | ID: mdl-38809826

RESUMO

PURPOSE: The current study compared temporal and spectral acoustic contrast between vowel segments produced by speakers with dysarthria across three speech tasks-interactive, solo habitual, and solo clear. METHOD: Nine speakers with dysarthria secondary to amyotrophic lateral sclerosis participated in the study. Each speaker was paired with a typical interlocutor over videoconferencing software. The speakers produced the vowels /i, ɪ, ɛ, æ/ in /h/-vowel-/d/ words. For the solo tasks, speakers read the stimuli aloud in both their habitual and clear speaking styles. For the interactive task, speakers produced a target stimulus for their interlocutor to select among the four possibilities. We measured the duration difference between long and short vowels, as well as the F1/F2 Euclidean distance between adjacent vowels, and also determined how well the vowels could be classified based on their acoustic characteristics. RESULTS: Temporal contrast between long and short vowels was higher in the interactive task than in both solo tasks. Spectral distance between adjacent vowel pairs was also higher for some pairs in the interactive task than the habitual speech task. Finally, vowel classification accuracy was highest in the interactive task. CONCLUSIONS: Overall, we found evidence that individuals with dysarthria produced vowels with greater acoustic contrast in structured interactions than they did in solo tasks. Furthermore, the speech adjustments they made to the vowel segments differed from those observed in solo speech.


Assuntos
Esclerose Lateral Amiotrófica , Disartria , Fonética , Acústica da Fala , Medida da Produção da Fala , Humanos , Disartria/etiologia , Disartria/fisiopatologia , Disartria/diagnóstico , Masculino , Feminino , Pessoa de Meia-Idade , Idoso , Esclerose Lateral Amiotrófica/complicações , Esclerose Lateral Amiotrófica/fisiopatologia , Inteligibilidade da Fala , Qualidade da Voz , Dados Preliminares , Espectrografia do Som , Fatores de Tempo , Idoso de 80 Anos ou mais , Acústica
7.
J Commun Disord ; 109: 106428, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-38744198

RESUMO

PURPOSE: This study examines whether there are differences in the speech of speakers with dysarthria, speakers with apraxia and healthy speakers in spectral acoustic measures during production of the central-peninsular Spanish alveolar sibilant fricative /s/. METHOD: To this end, production of the sibilant was analyzed in 20 subjects with dysarthria, 8 with apraxia of speech and 28 healthy speakers. Participants produced 12 sV(C) words. The variables compared across groups were the fricative's spectral amplitude difference (AmpD) and spectral moments in the temporal midpoint of fricative execution. RESULTS: The results indicate that individuals with dysarthria can be distinguished from healthy speakers in terms of the spectral characteristics AmpD, standard deviation (SD), center of gravity (CoG) and skewness, the last two in context with unrounded vowel, while no differences in kurtosis were detected. Participants with AoS group differ significantly from healthy speaker group in AmpD, SD and CoG and Kurtosis, the first one followed unrounded vowel and the latter two followed by rounded vowels. In addition, speakers with apraxia of speech group returned significant differences with respect to speakers with dysarthria group in AmpD, CoG and skewness. CONCLUSIONS: The differences found between the groups in the measures studied as a function of the type of vowel context could provide insights into the distinctive manifestations of motor speech disorders, contributing to the differential diagnosis between apraxia and dysarthria in motor control processes.


Assuntos
Apraxias , Disartria , Acústica da Fala , Humanos , Disartria/fisiopatologia , Disartria/etiologia , Apraxias/fisiopatologia , Masculino , Feminino , Pessoa de Meia-Idade , Adulto , Idoso , Fonética , Medida da Produção da Fala
9.
J Neural Transm (Vienna) ; 131(7): 791-797, 2024 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-38592459

RESUMO

INTRODUCTION: Hypokinetic dysarthria (HD) is a common motor speech symptom of Parkinson's disease (PD) which does not respond well to PD treatments. We investigated short-term effects of transcranial direct current stimulation (tDCS) on HD in PD using acoustic analysis of speech. Based on our previous studies we focused on stimulation of the right superior temporal gyrus (STG) - an auditory feedback area. METHODS: In 14 PD patients with HD, we applied anodal, cathodal and sham tDCS to the right STG using a cross-over design. A protocol consisting of speech tasks was performed prior to and immediately after each stimulation session. Linear mixed models were used for the evaluation of the effects of each stimulation condition on the relative change of acoustic parameters. We also performed a simulation of the mean electric field induced by tDCS. RESULTS: Linear mixed model showed a statistically significant effect of the stimulation condition on the relative change of median duration of silences longer than 50 ms (p = 0.015). The relative change after the anodal stimulation (mean = -5.9) was significantly lower as compared to the relative change after the sham stimulation (mean = 12.8), p = 0.014. We also found a correlation between the mean electric field magnitude in the right STG and improvement of articulation precision after anodal tDCS (R = 0.637; p = 0.019). CONCLUSIONS: The exploratory study showed that anodal tDCS applied over the auditory feedback area may lead to shorter pauses in a speech of PD patients.


Assuntos
Doença de Parkinson , Estimulação Transcraniana por Corrente Contínua , Humanos , Doença de Parkinson/terapia , Doença de Parkinson/complicações , Doença de Parkinson/fisiopatologia , Projetos Piloto , Masculino , Feminino , Idoso , Pessoa de Meia-Idade , Estudos Cross-Over , Disartria/etiologia , Disartria/terapia , Disartria/fisiopatologia , Fala/fisiologia , Resultado do Tratamento , Lobo Temporal/fisiopatologia
10.
Chest ; 165(4): e95-e100, 2024 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-38599764

RESUMO

CASE PRESENTATION: A 52-year-old woman with no significant medical history was referred to our hospital for expedited workup of progressive dysarthria and ataxia over the past year. Prior CT angiography of the head and neck showed no relevant neurologic findings but did reveal miliary lesions in the lung apices, which was later confirmed via dedicated CT chest scan (Fig 1). Review of systems was negative for any respiratory, constitutional, or rheumatologic symptoms, except for new xanthelasma-like lesions over her forehead. She previously had smoked with 20 pack-years and had no TB risk factors. MRI of the face showed a 21-mm mass within the left external temporal fascia. MRI of the head showed diffuse leptomeningeal enhancement, right frontal lobe enhancement, and cerebellar and brainstem T2/fluid-attenuated inversion recovery hyperintensity, which prompted her admission to hospital.


Assuntos
Ataxia Cerebelar , Disartria , Humanos , Feminino , Pessoa de Meia-Idade , Pulmão/patologia , Tomografia Computadorizada por Raios X , Pescoço
11.
IEEE J Transl Eng Health Med ; 12: 382-389, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-38606392

RESUMO

Acoustic features extracted from speech can help with the diagnosis of neurological diseases and monitoring of symptoms over time. Temporal segmentation of audio signals into individual words is an important pre-processing step needed prior to extracting acoustic features. Machine learning techniques could be used to automate speech segmentation via automatic speech recognition (ASR) and sequence to sequence alignment. While state-of-the-art ASR models achieve good performance on healthy speech, their performance significantly drops when evaluated on dysarthric speech. Fine-tuning ASR models on impaired speech can improve performance in dysarthric individuals, but it requires representative clinical data, which is difficult to collect and may raise privacy concerns. This study explores the feasibility of using two augmentation methods to increase ASR performance on dysarthric speech: 1) healthy individuals varying their speaking rate and loudness (as is often used in assessments of pathological speech); 2) synthetic speech with variations in speaking rate and accent (to ensure more diverse vocal representations and fairness). Experimental evaluations showed that fine-tuning a pre-trained ASR model with data from these two sources outperformed a model fine-tuned only on real clinical data and matched the performance of a model fine-tuned on the combination of real clinical data and synthetic speech. When evaluated on held-out acoustic data from 24 individuals with various neurological diseases, the best performing model achieved an average word error rate of 5.7% and a mean correct count accuracy of 94.4%. In segmenting the data into individual words, a mean intersection-over-union of 89.2% was obtained against manual parsing (ground truth). It can be concluded that emulated and synthetic augmentations can significantly reduce the need for real clinical data of dysarthric speech when fine-tuning ASR models and, in turn, for speech segmentation.


Assuntos
Percepção da Fala , Fala , Humanos , Interface para o Reconhecimento da Fala , Disartria/diagnóstico , Distúrbios da Fala
12.
Am J Speech Lang Pathol ; 33(4): 1639-1661, 2024 Jul 03.
Artigo em Inglês | MEDLINE | ID: mdl-38512013

RESUMO

PURPOSE: The purpose of this qualitative interview study was to identify themes regarding considerations in the usage of speech amplification device usage for people with Parkinson's disease (PD) and hypophonia from the perspective of speech-language pathologists (SLPs). METHOD: Eligible participants included SLPs currently practicing in the United States or Canada with experience working with clients with PD for at least 2 years. Ten SLPs participated in 60-min interviews conducted via Zoom. A semistructured interview guide was created prior to the interviews. The interviews were transcribed following their completion, and an iterative coding process was used to identify themes using thematic analysis. RESULTS: Three main themes were identified from the interviews. The first theme encapsulated how clinicians described amplification devices as a potential treatment tool, which highlighted the nuances that may impact selecting an amplification device as a treatment option such as increased hypophonia or dysarthria severity and cognitive decline. The second theme highlighted how device selection depends on the individual needs of the user. Individual client characteristics (such as disease symptoms and individual needs and preferences) may impact the choice of amplification device. The last theme outlined the importance of involving family members in all stages of device use and involving other health care team members on a case-by-case basis. CONCLUSIONS: The insights provided by the SLP participants help to understand the clinical decisions that are made when determining device candidacy, selecting a device, and evaluating device success. These insights can be used to improve research studies of augmentative management of hypophonia and guide more personalized management decisions.


Assuntos
Doença de Parkinson , Pesquisa Qualitativa , Patologia da Fala e Linguagem , Humanos , Patologia da Fala e Linguagem/métodos , Feminino , Masculino , Doença de Parkinson/complicações , Doença de Parkinson/terapia , Entrevistas como Assunto , Pessoa de Meia-Idade , Atitude do Pessoal de Saúde , Adulto , Disartria/terapia , Disartria/psicologia , Disartria/diagnóstico
13.
J Med Internet Res ; 26: e56417, 2024 Apr 25.
Artigo em Inglês | MEDLINE | ID: mdl-38509662

RESUMO

BACKGROUND: Dysarthria is a common poststroke speech disorder affecting communication and psychological well-being. Traditional speech therapy is effective but often poses challenges in terms of accessibility and patient adherence. Emerging smartphone-based therapies may offer promising alternatives for the treatment of poststroke dysarthria. OBJECTIVE: This study aimed to assess the efficacy and feasibility of smartphone-based speech therapy for improving speech intelligibility in patients with acute and early subacute poststroke dysarthria. This study also explored the impact of the intervention on psychological well-being, user experience, and overall feasibility in a clinical setting. METHODS: Participants were divided into 2 groups for this randomized, evaluator-blinded trial. The intervention group used a smartphone-based speech therapy app for 1 hour per day, 5 days per week, for 4 weeks, with guideline-based standard stroke care. The control group received standard guideline-based stroke care and rehabilitation. Speech intelligibility, psychological well-being, quality of life, and user acceptance were assessed using repeated measures ANOVA. RESULTS: In this study, 40 patients with poststroke dysarthria were enrolled, 32 of whom completed the trial (16 in each group). The intervention group showed significant improvements in speech intelligibility compared with the control group. This was evidenced by improvements from baseline (F1,30=34.35; P<.001), between-group differences (F1,30=6.18; P=.02), and notable time-by-group interactions (F1,30=6.91; P=.01). Regarding secondary outcomes, the intervention led to improvements in the percentage of correct consonants over time (F1,30=5.57; P=.03). In addition, significant reductions were noted in the severity of dysarthria in the intervention group over time (F1,30=21.18; P<.001), with a pronounced group effect (F1,30=5.52; P=.03) and time-by-group interaction (F1,30=5.29; P=.03). Regarding quality of life, significant improvements were observed as measured by the EQ-5D-3L questionnaire (F1,30=13.25; P<.001) and EQ-VAS (F1,30=7.74; P=.009) over time. The adherence rate to the smartphone-based app was 64%, with over half of the participants completing all the sessions. The usability of the app was rated high (system usability score 80.78). In addition, the intervention group reported increased self-efficacy in using the app compared with the control group (F1,30=10.81; P=.003). CONCLUSIONS: The smartphone-based speech therapy app significantly improved speech intelligibility, articulation, and quality of life in patients with poststroke dysarthria. These findings indicate that smartphone-based speech therapy can be a useful assistant device in the management of poststroke dysarthria, particularly in the acute and early subacute stroke stages. TRIAL REGISTRATION: ClinicalTrials.gov NCT05146765; https://clinicaltrials.gov/ct2/show/NCT05146765.


Assuntos
Disartria , Estudos de Viabilidade , Smartphone , Fonoterapia , Acidente Vascular Cerebral , Humanos , Disartria/terapia , Disartria/etiologia , Fonoterapia/métodos , Masculino , Feminino , Projetos Piloto , Pessoa de Meia-Idade , Acidente Vascular Cerebral/complicações , Idoso , Qualidade de Vida , Reabilitação do Acidente Vascular Cerebral/métodos , Aplicativos Móveis , Resultado do Tratamento
14.
Am J Speech Lang Pathol ; 33(3): 1390-1405, 2024 May.
Artigo em Inglês | MEDLINE | ID: mdl-38530396

RESUMO

PURPOSE: Changes in voice and speech are characteristic symptoms of Huntington's disease (HD). Objective methods for quantifying speech impairment that can be used across languages could facilitate assessment of disease progression and intervention strategies. The aim of this study was to analyze acoustic features to identify language-independent features that could be used to quantify speech dysfunction in English-, Spanish-, and Polish-speaking participants with HD. METHOD: Ninety participants with HD and 83 control participants performed sustained vowel, syllable repetition, and reading passage tasks recorded with previously validated methods using mobile devices. Language-independent features that differed between HD and controls were identified. Principal component analysis (PCA) and unsupervised clustering were applied to the language-independent features of the HD data set to identify subgroups within the HD data. RESULTS: Forty-six language-independent acoustic features that were significantly different between control participants and participants with HD were identified. Following dimensionality reduction using PCA, four speech clusters were identified in the HD data set. Unified Huntington's Disease Rating Scale (UHDRS) total motor score, total functional capacity, and composite UHDRS were significantly different for pairwise comparisons of subgroups. The percentage of HD participants with higher dysarthria score and disease stage also increased across clusters. CONCLUSION: The results support the application of acoustic features to objectively quantify speech impairment and disease severity in HD in multilanguage studies. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.25447171.


Assuntos
Doença de Huntington , Acústica da Fala , Medida da Produção da Fala , Humanos , Doença de Huntington/diagnóstico , Doença de Huntington/complicações , Masculino , Feminino , Pessoa de Meia-Idade , Adulto , Estudos de Casos e Controles , Idoso , Disartria/diagnóstico , Disartria/etiologia , Disartria/fisiopatologia , Análise de Componente Principal , Qualidade da Voz , Distúrbios da Fala/diagnóstico , Distúrbios da Fala/etiologia , Valor Preditivo dos Testes
15.
Front Immunol ; 15: 1308068, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-38524138

RESUMO

Background: Autoimmune nodopathy (AN) has emerged as a novel diagnostic category that is pathologically different from classic chronic inflammatory demyelinating polyneuropathy. Clinical manifestations of AN include sensory or motor neuropathies, sensory ataxia, tremor, and cranial nerve involvement. AN with a serum-positive contactin-1 (CNTN1) antibody usually results in peripheral nerve demyelination. In this study, we reported a rare case of AN with CNTN1 antibodies characterized by the presence of CNTN1 antibodies in both serum and cerebrospinal fluid, which is associated with cerebellar dysarthria. Methods: A 25-year-old man was admitted to our hospital due to progressive dysarthria with limb tremors. The patient was initially diagnosed with peripheral neuropathy at a local hospital. Three years after onset, he was admitted to our hospital due to dysarthria, apparent limb tremor, and limb weakness. At that time, he was diagnosed with spinocerebellar ataxia. Eight years post-onset, during his second admission, his condition had notably deteriorated. His dysarthria had evolved to typical distinctive cerebellar characteristics, such as tremor, loud voice, stress, and interrupted articulation. Additionally, he experienced further progression in limb weakness and developed muscle atrophy in the distal limbs. Magnetic resonance imaging (MRI), nerve conduction studies (NCS), and autoimmune antibody tests were performed. Results: The results of the NCS suggested severe demyelination and even axonal damage to the peripheral nerves. MRI scans revealed diffuse thickening of bilateral cervical nerve roots, lumbosacral nerve roots, cauda equina nerve, and multiple intercostal nerve root sheath cysts. Furthermore, anti-CNTN1 antibody titers were 1:10 in the cerebrospinal fluid (CSF) and 1:100 in the serum. After one round of rituximab treatment, the patient showed significant improvement in limb weakness and dysarthria, and the CSF antibodies turned negative. Conclusion: Apart from peripheral neuropathies, cerebellar dysarthria (central nervous system involvement) should not be ignored in AN patients with CNTN1 antibodies.


Assuntos
Disartria , Polirradiculoneuropatia Desmielinizante Inflamatória Crônica , Masculino , Humanos , Adulto , Disartria/complicações , Tremor/complicações , Contactina 1 , Ataxia
16.
Cereb Cortex ; 34(3)2024 03 01.
Artigo em Inglês | MEDLINE | ID: mdl-38466117

RESUMO

Speech disorders are associated with different degrees of functional and structural abnormalities. However, the abnormalities associated with specific disorders, and the common abnormalities shown by all disorders, remain unclear. Herein, a meta-analysis was conducted to integrate the results of 70 studies that compared 1843 speech disorder patients (dysarthria, dysphonia, stuttering, and aphasia) to 1950 healthy controls in terms of brain activity, functional connectivity, gray matter, and white matter fractional anisotropy. The analysis revealed that compared to controls, the dysarthria group showed higher activity in the left superior temporal gyrus and lower activity in the left postcentral gyrus. The dysphonia group had higher activity in the right precentral and postcentral gyrus. The stuttering group had higher activity in the right inferior frontal gyrus and lower activity in the left inferior frontal gyrus. The aphasia group showed lower activity in the bilateral anterior cingulate gyrus and left superior frontal gyrus. Across the four disorders, there were concurrent lower activity, gray matter, and fractional anisotropy in motor and auditory cortices, and stronger connectivity between the default mode network and frontoparietal network. These findings enhance our understanding of the neural basis of speech disorders, potentially aiding clinical diagnosis and intervention.


Assuntos
Afasia , Córtex Auditivo , Disfonia , Gagueira , Humanos , Disartria , Funções Verossimilhança , Distúrbios da Fala
17.
Pract Neurol ; 24(4): 310-312, 2024 Jul 16.
Artigo em Inglês | MEDLINE | ID: mdl-38423756

RESUMO

Paroxysmal dysarthria ataxia syndrome presents with recurrent, brief, stereotyped events of dysarthria, limb clumsiness, unsteady gait and vertigo or dizziness that can occur in association with lesions in the midbrain. We describe a case of a woman presenting with paroxysmal dysarthria and ataxia secondary to a midbrain lesion, treated successfully with carbamazepine.


Assuntos
Ataxia , Disartria , Humanos , Feminino , Disartria/etiologia , Ataxia/complicações , Ataxia/etiologia , Ataxia/tratamento farmacológico , Pessoa de Meia-Idade , Anticonvulsivantes/uso terapêutico , Carbamazepina/uso terapêutico
18.
Clin Rehabil ; 38(6): 802-810, 2024 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-38374687

RESUMO

OBJECTIVE: To identify and agree on what outcome domains should be measured in research and clinical practice when working with stroke survivors who have dysarthria. DESIGN: Delphi process, two rounds of an online survey followed by two online consensus meetings. SETTING: UK and Australia. PARTICIPANTS: Stroke survivors with experience of dysarthria, speech and language therapists/pathologists working in stroke and communication researchers. METHODS: Initial list of outcome domains generated from existing literature and with our patient and public involvement group to develop the survey. Participants completed two rounds of this survey to rate importance. Outcomes were identified as 'in', 'unclear' or 'out' from the second survey. All participants were invited to two consensus meetings to discuss these results followed by voting to identify critically important outcome domains for a future Core Outcome Set. All outcomes were voted on in the consensus meetings and included if 70% of meeting participants voted 'yes' for critically important. RESULTS: In total, 148 surveys were fully completed, and 28 participants attended the consensus meetings. A core outcome set for dysarthria after stroke should include four outcome domains: (a) intelligibility of speech, (b) ability to participate in conversations, (c) living well with dysarthria, (d) skills and knowledge of communication partners (where relevant). CONCLUSIONS: We describe the consensus of 'what' speech outcomes after stroke are valued by all stakeholders including those with lived experience. We share these findings to encourage the measurement of these domains in clinical practice and research and for future research to identify 'how' best to measure these outcomes.


Assuntos
Técnica Delphi , Disartria , Reabilitação do Acidente Vascular Cerebral , Acidente Vascular Cerebral , Humanos , Disartria/etiologia , Disartria/reabilitação , Acidente Vascular Cerebral/complicações , Feminino , Masculino , Avaliação de Resultados em Cuidados de Saúde , Pessoa de Meia-Idade , Austrália , Consenso , Idoso , Inquéritos e Questionários , Reino Unido
19.
Am J Speech Lang Pathol ; 33(3): 1236-1253, 2024 May.
Artigo em Inglês | MEDLINE | ID: mdl-38416062

RESUMO

PURPOSE: The purposes of this study were (a) to investigate adult listeners' perceptions of age and gender in typically developing children and children with dysarthria and (b) to identify predictors of their estimates among auditory-perceptual parameters and an acoustic measure of vocal pitch (F0). We aimed to evaluate the influence of dysarthria on the listeners' impressions of age and gender against the background of typical developmental processes. METHOD: In a listening experiment, adult listeners completed age and gender estimates of 144 typically developing children (3-9 years of age) and 25 children with dysarthria (5-9 years of age). The Bogenhausen Dysarthria Scales for Childhood Dysarthria (BoDyS-KiD) were applied to record speech samples and to complete auditory-perceptual judgments covering all speech subsystems. Furthermore, each child's mean F0 was determined from samples of four BoDyS-KiD sentences. RESULTS: Age estimates for the typically developing children showed a regression to the mean, whereas children with dysarthria were systematically underestimated in their age. The estimates of all children were predicted by developmental speech features; for the children with dysarthria, specific dysarthria symptoms had an additional effect. We found a significantly higher accuracy of gender attribution in the typically developing children than in the children with dysarthria. The prediction accuracy of the listeners' gender attribution in the preadolescent children by the included speech characteristics was limited. CONCLUSIONS: Children with dysarthria are more difficult to estimate for their age and gender than their typically developing peers. Dysarthria thus alters the auditory-perceptual impression of indexical speech features in children, which must be considered another facet of the communication disorder associated with childhood dysarthria.


Assuntos
Disartria , Acústica da Fala , Percepção da Fala , Medida da Produção da Fala , Humanos , Disartria/etiologia , Disartria/diagnóstico , Disartria/psicologia , Feminino , Masculino , Pré-Escolar , Criança , Fatores Etários , Linguagem Infantil , Fatores Sexuais , Adulto , Qualidade da Voz , Julgamento
20.
Artigo em Inglês | MEDLINE | ID: mdl-38306019

RESUMO

Objective: Language dysfunction is one of the most common cognitive impairments in amyotrophic lateral sclerosis (ALS). Although discourse capacities are essential for daily functioning, verbal expressive language has not been widely investigated in ALS. The existing research available suggests that discourse impairments are prevalent. This study investigates verbal expressive language in people living with ALS (plwALS) in contrast to healthy controls (HC).Methods: 64 plwALS and 49 age, gender and education-matched healthy controls were ask to describe the Cookie Theft Picture Task. The recordings were analyzed for discourse productivity, discourse content, syntactic complexity, speech fluency and verb processing. We applied the Bayesian hypothesis-testing framework, incorporating the effects of dysarthria, cognitive impairment status (CIS), and premorbid crystalline verbal IQ.Results: Compared to HC, plwALS only showed a single impairment: speech dysfluency. Discourse productivity, discourse content, syntactic complexity and verb processing were not impaired. Cognition and dysarthria exceeded the influence of verbal IQ for total words spoken and content density. Cognition alone seemed to explain dysfluency. Body-agent verbs were produced at even higher rates than other verb types. For the remaining outcomes, verbal IQ was the most decisive factor.Conclusions: In contrast to existing research, our data demonstrates no discernible impairment in verbal expressive language in ALS. What our findings show to be decisive is accounting for the influence of dysarthria, cognitive impairment status, and verbal IQ as variables on spontaneous verbal expressive language. Minor impairments in verbal expressive language appear to be influenced to a greater degree by executive dysfunctioning and dysarthria than by language impairment.


Assuntos
Esclerose Lateral Amiotrófica , Transtornos da Comunicação , Transtornos da Linguagem , Humanos , Teorema de Bayes , Disartria/etiologia , Idioma , Testes Neuropsicológicos
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