RESUMEN
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.
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PURPOSE: The primary objective of this study was to determine if speech and pause measures obtained using a passage reading task and timing measures from a monosyllabic diadochokinesis (DDK) task differ across speakers of Canadian French diagnosed with amyotrophic lateral sclerosis (ALS) presenting with and without bulbar symptoms, and healthy controls. The secondary objective was to determine if these measures can reflect the severity of bulbar symptoms. METHOD: A total of 29 Canadian French speakers with ALS (classified as bulbar symptomatic [n = 14] or pre-symptomatic [n = 15]) and 17 age-matched healthy controls completed a passage reading task and a monosyllabic DDK task (/pa/ and /ta/), for up to three follow-up visits. Measures of speaking rate, total duration, speech duration, and pause events were extracted from the passage reading recordings using a semi-automated speech and pause analysis procedure. Manual analysis of DDK recordings provided measures of DDK rate and variability. RESULT: Group comparisons revealed significant differences (p = < .05) between the symptomatic group and the pre-symptomatic and control groups for all passage measures and DDK rates. Only the DDK rate in /ta/ differentiated the pre-symptomatic and control groups. Repeated measures correlations revealed moderate correlations (rrm = > 0.40; p = < 0.05) between passage measures of total duration, speaking rate, speech duration, and number of pauses, and ALSFRS-R total and bulbar scores, as well as between DDK rate and ALSFRS-R total score. CONCLUSION: Speech and pause measures in passage and timing measures in monosyllabic DDK tasks might be suitable for monitoring bulbar functional symptoms in French speakers with ALS, but more work is required to identify which measures are sensitive to the earliest stages of the disease.
Asunto(s)
Esclerosis Amiotrófica Lateral , Habla , Humanos , Esclerosis Amiotrófica Lateral/complicaciones , Canadá , Medición de la Producción del Habla/métodos , LenguajeRESUMEN
PURPOSE: The primary aim of this study was to establish the reliability of candidate items as a step in the development of the Amyotrophic Lateral Sclerosis-Bulbar Dysfunction Index-Remote (ALS-BDI-Remote), a novel tool being developed for the detection and monitoring of bulbar signs and symptoms in remote settings. METHOD: The set of candidate items included 40 items covering three domains: cranial nerve examination, auditory-perceptual evaluation, and functional assessment. Forty-eight participants diagnosed with ALS and exhibiting a range of bulbar disease severity were included. Data collection for each participant took place on Zoom over three sessions. During Session 1, the participants were instructed to adjust their Zoom settings and to optimize their recording environment (e.g., lighting, background noise). Their cognition and eating were screened to determine their ability to follow instructions and their eligibility to perform the swallowing and chewing tasks. During Session 2, two speech-language pathologists (SLPs) administered the tool consecutively to determine the items' interrater reliability. During Session 3, one of the SLPs readministered the tool within 2 weeks of Session 1 to assess test-retest reliability. The reliability of each item was estimated using weighted kappa and the percentage of agreement. To be considered reliable, the items had to reach a threshold of 0.5 weighted kappa or 80% percentage agreement (if skewed distribution of the scores) for both interrater and test-retest reliability. RESULTS: In total, 33 of the 40 candidate items reached the reliability cutoff for both reliability analyses. All assessment domains included reliable items. Items requiring very good visualization of structures or movements were generally less reliable. CONCLUSIONS: This study resulted in the selection of reliable items to be included in the next version of the ALS-BDI-Remote, which will undergo psychometric evaluation (reliability, validity, and responsiveness analyses). Additionally, the results contributed to our understanding of the remote administration of SLP assessments for telehealth applications.
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Esclerosis Amiotrófica Lateral , Humanos , Esclerosis Amiotrófica Lateral/diagnóstico , Reproducibilidad de los Resultados , Examen Neurológico , Deglución , Índice de Severidad de la EnfermedadRESUMEN
PURPOSE: This study sought to determine whether clinically interpretable kinematic features extracted automatically from three-dimensional (3D) videos were correlated with corresponding perceptual clinical orofacial ratings in individuals with orofacial impairments due to neurological disorders. METHOD: 45 participants (19 diagnosed with motor neuron diseases [MNDs] and 26 poststroke) performed two nonspeech tasks (mouth opening and lip spreading) and one speech task (repetition of a sentence "Buy Bobby a Puppy") while being video-recorded in a standardized lab setting. The color video recordings of participants were assessed by an expert clinician-a speech language pathologist-on the severity of three orofacial measures: symmetry, range of motion (ROM), and speed. Clinically interpretable 3D kinematic features, linked to symmetry, ROM, and speed, were automatically extracted from video recordings, using a deep facial landmark detection and tracking algorithm for each of the three tasks. Spearman correlations were used to identify features that were significantly correlated (p value < .05) with their corresponding clinical scores. Clinically significant kinematic features were then used in the subsequent multivariate regression models to predict the overall orofacial impairment severity score. RESULTS: Several kinematic features extracted from 3D video recordings were associated with their corresponding perceptual clinical scores, indicating clinical validity of these automatically derived measures. Different patterns of significant features were observed between MND and poststroke groups; these differences were aligned with clinical expectations in both cases. CONCLUSIONS: The results show that kinematic features extracted automatically from simple clinical tasks can capture characteristics used by clinicians during assessments. These findings support the clinical validity of video-based automatic extraction of kinematic features.
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Enfermedades del Sistema Nervioso , Habla , Animales , Perros , Habla/fisiología , Algoritmos , Fenómenos Biomecánicos/fisiologíaRESUMEN
Impaired articulation (e.g., articulatory accuracy) and prosody (e.g., slow speech rate) are considered primary diagnostic criterions for apraxia of speech both in neurodegenerative and post-stroke contexts. The primary aim of this study was to investigate the ability of participants with primary progressive apraxia of speech (PPAOS), a neurodegenerative disease characterised by initially isolated progressive apraxia of speech, to increase speech rate and the interaction between articulatory accuracy and speech rate. The secondary aim was to investigate the effect of syllable frequency and structure on this interaction. Four speakers with PPAOS, and four sex- and age-matched healthy speakers (HS) read eight two-syllable words embedded two times in a ten-syllable carrier phrase. Syllable frequency and structure were manipulated for the first syllable of the target words and controlled for the second syllable. All sentences were produced at three different target speech rates (conditions): habitual, regular (five syllables/second), and fast (seven syllables/second). Prosodic measures for target words and sentences were computed based on acoustic analysis of speech rate. Articulatory measures for words and sentences were rated based on a perceptual assessment of articulatory accuracy. Results show slower speech rate and reduced articulatory accuracy in speakers with PPAOS compared to HS. Results suggest that speakers with PPAOS also have limited ability to increase their speech rate. Finally, results suggest that articulatory complexity influences speech rate but that the cost of speech rate increase on articulatory accuracy varies greatly across speakers with PPAOS and is not necessarily related to the extent of the increase when measured in a highly structured sentence production task. Theoretical and clinical implications of these findings are discussed.
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Apraxias , Enfermedades Neurodegenerativas , Apraxias/diagnóstico , Humanos , Lenguaje , Habla , Medición de la Producción del HablaRESUMEN
The differential diagnosis of primary progressive aphasia (PPA) is challenging due to overlapping clinical manifestations of the different variants of the disease. This is particularly true for the logopenic variant of PPA (lvPPA), in which such overlap was reported with regard to impairments in repetition abilities. In this study, four individuals with lvPPA underwent standard neuropsychological and language assessments. The influence of psycholinguistic variables on their performance of in word, nonword and sentence repetition tasks was also specifically explored. Some level of heterogeneity was found in cognitive functions and in language. The four participants showed impairment in sentence repetition in which their performance was negatively affected by semantic reversibility and syntactic complexity. This study supports the heterogeneity of lvPPA with respect to the cognitive and linguistic status of participants. It also shows that sentence repetition is influenced not only by length, but also by semantic reversibility and syntactic complexity, two psycholinguistic variables known to place additional demands on phonological working memory.
O diagnóstico diferencial da afasia progressiva primária (APP) é desafiador devido às sobreposições das manifestações clínicas das diferentes variantes da doença. Isso é particularmente verdadeiro para a variante logopênica do APP (APPlg), em que tal sobreposição foi relatada em relação à deficiências nas habilidades de repetição. No presente estudo, quatro indivíduos com APPlg foram submetidos à avaliações neuropsicológica e de linguagem. A influência de variáveis psicolinguísticas em seu desempenho de palavras, não-palavras e tarefas de repetição de frases também foi especificamente explorada. Certo nível de heterogeneidade foi encontrado nas funções cognitivas e na linguagem. Os quatro participantes apresentaram prejuízo na repetição de frases em que seu desempenho foi afetado negativamente pela reversibilidade semântica e complexidade sintática. O estudo apoia a heterogeneidade do APPlg no que diz respeito ao status cognitivo e linguístico dos participantes. Mostra também que a repetição de sentenças é influenciada não apenas pelo comprimento, mas também pela reversibilidade semântica e complexidade sintática, duas variáveis psicolinguísticas conhecidas por colocarem demandas adicionais na memória operacional fonológica.
RESUMEN
ABSTRACT. The differential diagnosis of primary progressive aphasia (PPA) is challenging due to overlapping clinical manifestations of the different variants of the disease. This is particularly true for the logopenic variant of PPA (lvPPA), in which such overlap was reported with regard to impairments in repetition abilities. In this study, four individuals with lvPPA underwent standard neuropsychological and language assessments. The influence of psycholinguistic variables on their performance of in word, nonword and sentence repetition tasks was also specifically explored. Some level of heterogeneity was found in cognitive functions and in language. The four participants showed impairment in sentence repetition in which their performance was negatively affected by semantic reversibility and syntactic complexity. This study supports the heterogeneity of lvPPA with respect to the cognitive and linguistic status of participants. It also shows that sentence repetition is influenced not only by length, but also by semantic reversibility and syntactic complexity, two psycholinguistic variables known to place additional demands on phonological working memory.
RESUMO. O diagnóstico diferencial da afasia progressiva primária (APP) é desafiador devido às sobreposições das manifestações clínicas das diferentes variantes da doença. Isso é particularmente verdadeiro para a variante logopênica do APP (APPlg), em que tal sobreposição foi relatada em relação à deficiências nas habilidades de repetição. No presente estudo, quatro indivíduos com APPlg foram submetidos à avaliações neuropsicológica e de linguagem. A influência de variáveis psicolinguísticas em seu desempenho de palavras, não-palavras e tarefas de repetição de frases também foi especificamente explorada. Certo nível de heterogeneidade foi encontrado nas funções cognitivas e na linguagem. Os quatro participantes apresentaram prejuízo na repetição de frases em que seu desempenho foi afetado negativamente pela reversibilidade semântica e complexidade sintática. O estudo apoia a heterogeneidade do APPlg no que diz respeito ao status cognitivo e linguístico dos participantes. Mostra também que a repetição de sentenças é influenciada não apenas pelo comprimento, mas também pela reversibilidade semântica e complexidade sintática, duas variáveis psicolinguísticas conhecidas por colocarem demandas adicionais na memória operacional fonológica.
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Humanos , Afasia Progresiva Primaria , Trastorno Específico del Lenguaje , Psicolingüística , Diagnóstico DiferencialRESUMEN
Purpose This study aimed to track changes in acoustical and perceptual features of motor speech in patients with phonetic and prosodic primary progressive apraxia of speech (PPAOS) in Québec French over an 18-month period. Method A prospective multiple-case series with multiple testing periods, including four participants with a diagnosis of PPAOS, was conducted. Participants were 0.5-4 years postonset of disease at baseline. They underwent comprehensive motor speech and language assessments and cognitive screening every 6 months for up to 18 months. Acoustical and perceptual analyses of motor speech were conducted. Results Results showed a considerable impairment in motor speech abilities for patients with PPAOS at all time points and a significant decrease in performance for almost all articulatory and prosodic measures over time. Passage reading and diadochokinesis seemed particularly promising for the tracking of changes in PPAOS motor speech characteristics and PPAOS classification. Quantifying length of speech runs made it possible to distinguish phonetic from prosodic PPAOS. Finally, the patients who evolved to phonetic PPAOS developed aphasia, and the two with prosodic PPAOS showed greater motor symptoms such as unequivocal dysarthria. Conclusion This study extends the growing literature on PPAOS and its subtypes by describing specific changes in articulatory and prosodic abilities over a period of at least 6 months, which are important for the diagnosis and management of PPAOS.
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Afasia Progresiva Primaria , Apraxias , Apraxias/diagnóstico , Humanos , Estudios Prospectivos , Quebec , HablaRESUMEN
BACKGROUND: The term progressive apraxia of speech (PAOS) is used to describe speakers presenting with isolated or dominant apraxia of speech in the context of a neurodegenerative syndrome, including primary progressive apraxia of speech (PPAOS) and dominant progressive apraxia of speech (DAOS), respectively. Its motor speech profile has been increasingly explored in the last decade, but description remains vague and very English oriented, although the effect of speakers' language on motor speech phenotypes is increasingly recognized. Although some studies suggest that speakers presenting with isolated PAOS (PPAOS) versus dominant PAOS with concomitant aphasia (DAOS) should be differentiated, distinct characteristics of the two presentations are unclear. Furthermore, a careful description of their clinical presentation in languages other than English is required. AIMS: To describe the motor speech characteristics of Quebec French-speaking participants with prominent PAOS and to explore the communication profile of those presenting more specifically with isolated PAOS (PPAOS), and with dominant PAOS and concomitant aphasia (DAOS). METHODS & PROCEDURES: A thorough effort to recruit all speakers presenting with PAOS in the larger population areas of the province of Quebec was conducted over a 3-year span. A total of nine participants with PAOS (pwPAOS; PPAOS = 5, DAOS = 4) underwent a comprehensive language and motor speech assessment, and a cognitive screening. Their performance was compared with 30 matched healthy controls. OUTCOMES & RESULTS: As a group, pwPAOS differed from healthy speakers on all acoustic and perceptual measures. The PPAOS and PAOS subgroups were similar on several measures, but participants from the PPAOS subgroup tended to perform better on articulatory measures and maximum speech rate tasks. CONCLUSIONS & IMPLICATIONS: This study provides an in-depth analysis of motor speech characteristics of PAOS in Quebec French speakers and adds further evidence for the differentiation of PPAOS and DAOS. Combining simple perceptual and acoustic analyses represent a promising approach to distinguish the two variants and identify treatment targets. What this paper adds What is already known on this subject Progressive apraxia of speech (PAOS) is a neurodegenerative syndrome characterized by progressive and initially isolated or dominant apraxia of speech (primary progressive apraxia of speech [PPAOS] and dominant progressive apraxia of speech [DAOS], respectively). Studies mostly report articulatory and prosodic deficits in PAOS, but concomitant deficits such as dysarthria and executive dysfunction are also reported. The description of motor speech skills in PAOS remains vague and English-oriented. Studies suggest that speakers presenting with isolated PAOS vs dominant PAOS with concomitant aphasia should be differentiated, but distinct characteristics of the two presentations are unclear. What this study adds to existing knowledge To the best of the authors' knowledge, this study is the first to report transversal data of Quebec-French participants with PPAOS and DAOS. Moreover, this study is a first step towards identifying potential characteristics that could facilitate the diagnosis of PPAOS and DAOS in Quebec French. It makes a significant contribution to our understanding of progressive apraxia of speech in different cultural languages. What are the potential or actual clinical implications of this work? This study also initiates the search for sensitive tasks for the diagnosis of those speakers (which is an important process), in addition to identifying the core characteristics of PAOS, DAOS, and PPAOS in the development of an assessment battery for this population.
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Afasia Progresiva Primaria , Apraxias , Apraxias/diagnóstico , Humanos , Lenguaje , Quebec , HablaRESUMEN
Primary progressive apraxia of speech (PPAoS) is a neurodegenerative syndrome characterized by speech apraxia at its onset; as it progresses, it often evolves into total mutism. Even though this syndrome is increasingly recognized, its early differential diagnostic is still complex. The objective of this study was to illustrate why a fine evaluation of speech and language is essential for the differential diagnosis of PPAoS. This longitudinal case study presents the progression of a PPAoS patient over a period of 5 years. Periodic neurological and speech-language assessments were carried out to follow the progression of neurological, memory, language and speech symptoms. The different diagnostic labels established over time were also reported. The evolution of the patient's communication profile was characterized by a preservation of language components and episodic memory, in parallel with a progressive deterioration of speech which gradually reduced intelligibility, and was associated with signs of spasticity, resulting in a complete anarthria. This case study sheds light upon the evolution of a patient with PPAoS. A better understanding of the clinical profile and progression of PPAoS is necessary in order to improve early diagnosis and adequate care for these patients.
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Apraxias/diagnóstico , Diagnóstico Precoz , Trastornos del Lenguaje/diagnóstico , Comunicación , Progresión de la Enfermedad , Humanos , Trastornos del Habla/diagnósticoRESUMEN
Language disorders following subcortical non-thalamic stroke show great variability across literature and a well-defined profile in these aphasic disturbances is yet to be established. The lack of recent and consistent literature on the subject complicates the management of subcortical aphasia. The aim of this study is to review the literature describing oral language disturbances following subcortical non-thalamic stoke affecting the basal ganglia and the surrounding white matter. A review of the literature of three databases (PubMed, PsycNet and LLBA), identifying research articles from 1997 to 2015, was completed. The quality of the selected studies was assessed using the Checklist for the assessment of methodological quality. Twenty-two articles met criteria for review and oral language assessment data were extracted for 114 subjects. The results suggest a predominance of deficits in more complex and demanding language levels (ex. discourse, syntax) and in language production (vs comprehension). Rapid recovery is expected, especially for lexical-semantic and receptive deficits. These findings show the importance of a complete oral language evaluation after subcortical stoke and provide recent data relative to expected deficits and recovery to guide clinicians in the management of these patients. They also suggest that a descriptive approach of the deficits may be more efficient and accurate than the use of a traditional classification of aphasia.