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OBJECTIVES: The aim was to describe the acoustic, auditory-perceptive, and subjective voice changes under the Lombard effect (LE) in adductor laryngeal dystonia (AdLD) patients. METHODS: Subjective perception of vocal effort (OMNI Vocal Effort Scale OMNI-VES), Maximum Phonation Time (MPT), and the perceptual severity of dysphonia (GRBAS scale) were assessed in condition of stillness and under LE in 10 AdLD patients and in 10 patients with typical voice. Speakers were asked to produce the sustained vowel /a/ and to read a phonetically balanced text aloud. Using the PRAAT software, the following acoustic parameters were analyzed: Mean Pitch (Hz), Minimum and Maximum Intensity (dB), the Fraction of Locally Unvoiced Frames, the Number of Voice Breaks, the Degree of Voice Breaks (%), the Cepstral Peak Prominence-Smoothed (CPPS) (dB). RESULTS: Under LE, the AdLD group showed a decrease of both G and S parameters of GRBAS and subjective effort, mean MPT increased significantly; in the controls there were no significant changes. In both groups under LE, pitch and intensity of the sustained vowel /a/ significantly increased consistently with LE. In the AdLD group the mean gain of OMNI-VES score and the mean gain of each parameter of the speech analysis were significantly greater than the controls' ones. CONCLUSION: Auditory feedback deprivation obtained under LE improves subjective, perceptual-auditory, and acoustics parameters of AdLD patients. These findings encourage further research to provide new knowledge into the role of the auditory system in the pathogenesis of AdLD and to develop new therapeutic strategies. LEVEL OF EVIDENCE: 4 Laryngoscope, 134:3754-3760, 2024.
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Calidad de la Voz , Humanos , Proyectos Piloto , Masculino , Femenino , Persona de Mediana Edad , Calidad de la Voz/fisiología , Adulto , Anciano , Enfermedades de la Laringe/fisiopatología , Enfermedades de la Laringe/complicaciones , Acústica del Lenguaje , Distonía/fisiopatología , Fonación/fisiología , Disfonía/fisiopatologíaRESUMEN
OBJECTIVE: Diagnosis of adductor laryngeal dystonia (AdLD) is challenging as it mimics voice features of other voice disorders. This could lead to misdiagnosis (or delayed diagnosis) and ineffective treatments of AdLD. This paper develops automated measurements of glottal attack time (GAT) and glottal offset time (GOT) from high-speed videoendoscopy (HSV) in connected speech as objective measures that can potentially facilitate the diagnosis of this disorder in the future. METHODS: HSV data were recorded from vocally normal adults and patients with AdLD during the reading of the "Rainbow Passage" and six CAPE-V (Consensus Auditory-Perceptual Evaluation of Voice) sentences. A deep learning framework was designed and trained to segment the glottal area and detect the vocal fold edges in the HSV dataset. This automated framework allowed us to automatically measure and quantify the GATs and GOTs for the participants. Accordingly, a comparison was held between the obtained measurements among vocally normal speakers and those with AdLD. RESULTS: The automated framework was successfully developed and able to accurately segment the glottal area/edges. The precise automated measurements of GAT and GOT revealed minor, nonsignificant differences compared to the results of manual analysis-showing a strong correlation between the measures by the automated and manual methods. The results showed significant differences in the GAT values between the vocally normal subjects and AdLD patients, with larger variability in both the GAT and GOT measures in the AdLD group. CONCLUSIONS: The developed automated approach for GAT and GOT measurement can be valuable in clinical practice. These quantitative measurements can be used as meaningful biomarkers of the impaired vocal function in AdLD and help its differential diagnosis in the future.
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BACKGROUND: The pathophysiology of adductor laryngeal dystonia (AdLD) remains unknown; however, there is growing evidence that dystonia is associated with disruptions in the inhibitory regulation of sensorimotor cortical areas. Using functional MRI (fMRI) and transcranial magnetic stimulation (TMS) complementarily, we previously demonstrated an overly activated laryngeal motor cortex and revealed correlations between blood-oxygen-level dependent (BOLD) activation and intracortical inhibition in a phonation (dystonia-related) task in adductor laryngeal dystonia (AdLD). OBJECTIVE: Here, we aimed to characterize the brain-based findings in the primary motor cortex (M1) during a dystonia-unrelated (finger tapping) task in AdLD and controls (CTL). METHODS: We examined the between-group differences in task-dependent BOLD activation and intracortical inhibition, measured by the TMS-evoked cortical silent period (cSP), in the M1. The correlations between fMRI and TMS responses were assessed. RESULTS: There is more broadly dispersed BOLD activation, not confined to the hand motor cortex, and reduced intracortical inhibition in AdLD compared to CTL. Further, there are more positive correlations between cSP and BOLD activation in a task unrelated to dystonic symptoms in AdLD compared with CTL. This is in contrast to our previous work that demonstrated fewer positive correlations in AdLD during a dystonic phonation task. CONCLUSIONS: In unaffected musculature activation, there is dispersed BOLD activation that is correlated with intracortical inhibition suggesting a possible compensatory strategy in the non-dystonic muscles.
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Disfonía , Distonía , Trastornos Distónicos , Corteza Sensoriomotora , Humanos , Distonía/diagnóstico por imagen , Estimulación Magnética Transcraneal , Potenciales Evocados Motores/fisiologíaRESUMEN
OBJECTIVES: Differential diagnosis for adductor laryngeal dystonia (AdLD) is often carried out by comparing symptom expression during sentences with either all voiced or voiced and voiceless consonants. However, empirical research examining the effects of phonetic context on symptoms is sparse. The purpose of this study was to examine whether symptom probabilities varied across voiced speech segments in an all-voiced sentence, and whether this variability was systematic with respect to phonetic features. METHODS: Eighteen speakers with AdLD read aloud a sentence comprised entirely of voiced speech sounds. Speech segment boundaries and AdLD symptoms (phonatory breaks, frequency shifts, and creak) were labeled separately, and speech segments were coded as symptomatic or asymptomatic based on their temporal overlap. Generalized linear mixed effects models with a binomial outcome variable were used to compare the probability of symptom expression across: 1) all speech segments in the sentence, and 2) four speech sound classes (vowels, approximants, nasals, and obstruents). RESULTS: Significant symptom variability was found across voiced speech segments in the sentence. Furthermore, the estimated probability of a symptom occurring on vowels and approximants was significantly greater than that of nasals and obstruents. CONCLUSION: These results indicate that AdLD symptoms are not uniformly distributed across voiced speech segments with systematic variation across speech sound classes.To explain these findings, future work should investigate how the complex interactions between the vocal tract articulators and glottal configurations may influence symptom expression in this population.
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PURPOSE: The effects of neuromodulation are virtually unexplored in adductor laryngeal dystonia (AdLD), a disorder characterized by involuntary contraction of intrinsic laryngeal muscles. Recent findings indicated that intracortical inhibition is reduced in people with AdLD. Low-frequency repetitive transcranial magnetic stimulation (rTMS) induces prolonged intracortical inhibition, but the effects in AdLD are unexplored. This pilot and feasibility study aimed to examine the safety, feasibility, and effects of a single session 1 Hz rTMS over the laryngeal motor cortex (LMC) in people with AdLD and healthy individuals. METHODS: The stimulation location was individualized and determined through TMS-evoked responses in the thyroarytenoid muscles using fine-wire electrodes. 1200 pulses of 1 Hz rTMS were delivered to the left LMC in two groups: Control (n = 6) and AdLD (n = 7). Tolerance, adverse effects, intracortical inhibition, and voice recordings were collected immediately before and after rTMS. Voice quality was assessed with acoustic-based and auditory-perceptual measures. RESULTS: All participants tolerated the procedures, with no unexpected adverse events or worsening of symptoms. No significant effects on intracortical inhibition were observed. In the AdLD group, there was a large-effect size after rTMS in vocal perturbation measures and a small-effect size in decreased phonatory breaks. CONCLUSIONS: One rTMS session over the LMC is safe and feasible, and demonstrated trends of beneficial effects on voice quality and phonatory function in AdLD. These preliminary findings support further investigation to assess clinical benefits in a future randomized sham-controlled trial. CLINICALTRIALS.GOV: NCT02957942, registered on November 8, 2016.