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Objective: Differentiating adductor laryngeal dystonia (ADLD) and primary muscle tension dysphonia (pMTD) can be challenging. Unlike pMTD, ADLD is described as "task-dependent" with voiced phonemes purportedly provoking greater sign expression than voiceless phonemes. We evaluated the ability of two automated acoustic measures, the Cepstral Spectral Index of Dysphonia (CSID) and creak, to detect task dependency and to discriminate ADLD and pMTD. Method: CSID, % creak, and listener ratings of dysphonia severity were obtained from audio recordings of patients with ADLD (n = 29) or pMTD (n = 33) reading two sentences loaded with either voiced or voiceless phonemes. Results: Group × Sentence Type interaction effects confirmed that both "normalized" CSID and % creak detected task-dependent sign expression in ADLD (i.e., worse symptoms on the voiced- vs. voiceless-loaded sentence). However, a stepwise binary logistic regression analysis with group (ADLD vs. pMTD) as the dependent variable and % creak and normalized CSID variables (voiced, voiceless, and voiced vs. voiceless difference) as covariates revealed that the normalized CSID voiceless-laden sentence z score was the only significant predictor of group membership. Estimates of diagnostic precision from the normalized CSID voiceless sentence z scores were superior to % creak or listener ratings. Finally, the CSID possessed the strongest correlations with listener severity ratings regardless of group or sentence type. Conclusions: Although both normalized CSID and % creak detected taskdependent performance as a distinguishing feature of ADLD, a CSID profile wherein (a) the voiceless sentence z score was less severe than the voiced sentence and (b) the normalized voiceless sentence z score was within approximately 2 SDs (or less) of typical expectations provided the best estimates of diagnostic precision. Automated acoustic measures such as the CSID and creak provide useful information to objectively discriminate ADLD and pMTD. [ABSTRACT FROM AUTHOR]

Objectives: Maximum phonation time is a simple test used to assess glottic competency. Our objective was to evaluate any correlation between maximum phonation time and spasmodic dysphonia as adductor spasmodic dysphonia and abductor spasmodic dysphonia have an adductor and abductor overdrive, respectively. Methods: A 3-year data-review was performed for patients diagnosed with adductor spasmodic dysphonia, abductor spasmodic dysphonia and mixed spasmodic dysphonia. Maximum phonation time was noted on the first visit and compared with a control group. Results: Average maximum phonation time in adductor spasmodic dysphonia, abductor spasmodic dysphonia and control group was 25 seconds, 9 seconds and 16 seconds. A significant difference was found for adductor spasmodic dysphonia and abductor spasmodic dysphonia. A receiver operating characteristic curve analysis between adductor spasmodic dysphonia and control groups showed a positive predictive value of 81.3 per cent, negative predictive value of 83.9 per cent, sensitivity of 79.6 per cent and specificity of 85.2 per cent. Level of evidence = 4. Conclusion: We recommend that maximum phonation time be added to the diagnostic armamentarium of spasmodic dysphonia. This correlation between maximum phonation time and spasmodic dysphonia has not been previously published. [ABSTRACT FROM AUTHOR]

Purpose: Laryngeal dystonia (LD) is a focal dystonia affecting laryngeal musculature with no known etiology or cure. The present study evaluated the sociodemographic and clinical features of patients diagnosed with LD. Materials and Methods: All patients diagnosed with LD at our University Hospital's Ear, Nose, and Throat Department between January 2017 and July 2023 were retrospectively analyzed. The study included 43 patients. Results: Out of the 43 patients, 19 (44%) were male. At the time of diagnosis, the mean age of the patients was 35.1 years (ranging from 17 to 65 years). The mean elapsed time between the first symptom onset and the first diagnosis was 49.2 months (min. 4 months, max. 240 months). Of the participants, 94% had adductor-type LD. None of the patients had a family history of LD. Of the patients, 9 (20%) experienced a life-altering event or trauma just before the onset of symptoms. All patients who consumed alcohol reported symptom relief with alcohol intake. A total of 67.6% of patients stated that their symptoms were triggered by stress. All of our patients received at least one Botulinum toxin injection, with an average of 2.75 dosages per patient. Conclusion: The gender distribution was approximately equitable between males and females. There was a tendency for men to receive a diagnosis earlier than women following the manifestation of symptoms. A significant number of patients associate the emergence of their symptoms with a stressful event or traumatic experience. This study represents the initial investigation into the sociodemographic characteristics of patients within the Turkish population. [ABSTRACT FROM AUTHOR]

Background: Prior studies have indicated that female individuals outnumber male individuals for certain types of dystonia. Few studies have addressed factors impacting these sex differences or their potential biological mechanisms. Objectives: To evaluate factors underlying sex differences in the dystonias and explore potential mechanisms for these differences. Methods: Data from individuals with various types of dystonia were analyzed in relation to sex. Data came from two different sources. One source was the Dystonia Coalition database, which contains predominantly idiopathic adult‐onset focal and segmental dystonias. The second source was the MDSGene database, which contains predominantly early‐onset monogenic dystonias. Results: The 3222 individuals from the Dystonia Coalition included 71% female participants and 29% male participants for an overall female‐to‐male ratio (F:M) of 2.4. This ratio varied according to body region affected and whether dystonia was task‐specific. The female predominance was age‐dependent. Sex did not have a significant impact on co‐existing tremor, geste antagoniste, depression or anxiety. In the 1377 individuals from the MDSGene database, female participants outnumbered male participants for some genes (GNAL, GCH1, and ANO3) but not for other genes (THAP1, TH, and TOR1A). Conclusions: These results are in keeping with prior studies that have indicated female individuals outnumber male individuals for both adult‐onset idiopathic and early onset monogenic dystonias. These results extend prior observations by revealing that sex ratios depend on the type of dystonia, age, and underlying genetics. [ABSTRACT FROM AUTHOR]

Background: Laryngeal dystonia is a task-specific focal dystonia of laryngeal muscles that impairs speech and voice production. At present, there is no cure for LD. The most common therapeutic option for patients with LD involves Botulinum neurotoxin injections. Objective: Provide empirical evidence that non-invasive vibro-tactile stimulation (VTS) of the skin over the voice box can provide symptom relief to those affected by LD. Methods: Single-group 11-week randomized controlled trial with a crossover between two dosages (20 min of VTS once or 3 times per week) self-administered in-home in two 4-week blocks. Acute effects of VTS on voice and speech were assessed in-lab at weeks 1, 6 and 11. Participants were randomized to receive either 40 Hz or 100 Hz VTS. Main outcome measures: Primary: smoothed cepstral peak prominence (CPPS) of the voice signal to quantify voice and speech abnormalities, and perceived speech effort (PSE) ranked by participants as a measure of voice effort (scale 1-10). Secondary: number of voice breaks during continuous speech, the Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) inventory as a measure of overall disease severity and the Voice Handicap Index 30-item self report. Results: Thirty-nine people with a confirmed diagnosis of adductor-type LD (mean [SD] age, 60.3 [11.3] years; 18 women and 21 men) completed the study. A single application of VTS improved voice quality (median CPPS increase: 0.41 dB, 95% CI [0.20, 0.61]) and/or reduced voice effort (PSE) by at least 30% in up to 57% of participants across the three study visits. Effects lasted from less than 30 min to several days. There was no effect of dosage and no evidence that the acute therapeutic effects of VTS increased or decreased longitudinally over the 11-week study period. Both 100 and 40 Hz VTS induced measurable improvements in voice quality and speech effort. VTS induced an additional benefit to those receiving Botulinum toxin. Participants, not receiving Botulinum treatment also responded to VTS. Conclusion: This study provides the first systematic empirical evidence that the prolonged use of laryngeal VTS can induce repeatable acute improvements in voice quality and reductions of voice effort in LD. Clinical trial registration: ClinicalTrials.gov ID: NCT03746509. [ABSTRACT FROM AUTHOR]

The article discusses a case report of a 45-year-old male patient with abductor laryngeal dystonia (ABLD) who underwent pallidal deep brain stimulation (DBS) surgery. ABLD is a rare subtype of laryngeal dystonia characterized by breathy breaks during speech. The patient experienced immediate and remarkable efficacy of DBS, with full recovery from symptoms without recurrence. The study highlights the potential of pallidal DBS as a highly effective treatment for ABLD, offering new insights into the management of this challenging condition. [Extracted from the article]

A variety of surgical treatment options exist for adductor spasmodic dysphonia (ADSD) with selective adductor recurrent laryngeal nerve denervation and reinnervation (SLAD‐R) being one of the more popular. We present a case of bilateral vocal fold paralysis (BVFP) for SLAD‐R resulting in the need for total laryngectomy. We suggest BVFP is more common than reported and that we all must insure optimal long term follow up of our surgical patients. Laryngoscope, 134:4582–4584, 2024 [ABSTRACT FROM AUTHOR]

Introduction: Adductor spasmodic dysphonia (ADSD) is characterized by involuntary laryngeal muscle spasms. Due to the lack of a quantitative evaluation method, most measurements have demonstrated difficulty in validity and reliability for diagnosing ADSD. This study aimed to establish a novel indicator for ADSD and determine its diagnostic effects. Methods: We investigated 98 voice samples from 49 patients with ADSD and 49 healthy participants. A sustained vowel was recorded by a high-definition audio recorder. Voice samples underwent regular acoustic evaluation and a novel global dimension (GD) method. GD, Jitter, Shimmer, HNR, Frequency shift, and CPPS were measured for both groups. Results: Statistical analysis revealed that the GD method effectively differentiated ADSD patients from healthy participants (p < 0.001, D′>0.8). Subsequent multiclass receiver operating characteristic analysis demonstrated that GD possessed the most significant classification accuracy (area under the curve = 0.988) compared with other acoustic parameters. Conclusion: GD was an effective metric for objective differentiation between ADSD patients and healthy participants. This metric could assist clinicians in the diagnosis of ADSD patients. [ABSTRACT FROM AUTHOR]

Adenylyl cyclase 5 (ADCY5)-related dyskinesia is a rare disorder characterized by early-onset paroxysmal choreoathetosis, dystonia, myoclonus, or a combination thereof, primarily involving the limbs, face, and neck. Other core clinical features include nocturnal ballistic bouts and facial dyskinesias, often followed by remission during adolescence, as well as axial hypotonia. Any mixed movement disorder accompanied by the aforementioned core features should prompt genetic testing for ADCY5 mutation. We report the case of a 31-year man from North India with a childhood-onset history of clumsy, random, and jerky body and facial movements, interfering with his speech and object-handling and walking-related functional abilities. He improved symptomatically with zonisamide and was offered caffeine as a maintenance option. Here, we review the literature on this entity, while reporting this first case of ADCY5-related dyskinesias from North India. The phenotypic spectrum linked to ADCY5 mutations has significantly broadened since its initial description in a family with "familial dyskinesia and facial myokymia." This phenotypic variability could be responsible for a proportion of "idiopathic" hyperkinetic movement disorders. Gaining a better understanding of its clinical manifestations helps in identifying complex or uncommon cases more effectively. [ABSTRACT FROM AUTHOR]

Objectives: There is an absence of data in the literature regarding methods to improve the patient experience during the performance of awake in‐office laryngeal injections. This study sought to evaluate whether the use of local anesthetic or a vibrating instrument decreased overall pain experienced by patients with laryngeal dystonia, frequently referred to as spasmodic dysphonia (SD), undergoing transcervical botulinum toxin injections. Methods: This was an unblinded, prospective randomized control trial with a crossover design where each patient received transcutaneous transcricothyroid injection of botulinum toxin with alternating use of no anesthesia, local anesthesia (2% lidocaine in 1:100,000 epinephrine), and vibrating instrument in three consecutive laryngeal injections to treat adductor SD. Patients were randomized to the order they received these treatments. Patients measured pain on a 0–10 visual analogue scale (VAS) and selected their preferred technique after receiving all three analgesic modalities. Results: Thirty‐two patients completed the study. There was no statistically significant difference in pain between the three analgesic techniques (p = 0.38). The most preferred analgesic technique was the vibrating wand (44% (14/32)). Lidocaine was the second most preferred (37% (12/32)) and 19% (6/32) of patients preferred nothing. When combining the wand and nothing groups, 63% of patients preferred one of these two methods (95% exact CI: 44%–79%). Conclusion: There was no statistically significant difference in median pain experienced by patients during laryngeal botulinum toxin injection between these different analgesic modalities. More than half of the patients selected a preference for a technique that did not include lidocaine. This data supports individualization of analgesia during transcutaneous laryngeal injections. Level of Evidence: 2 Laryngoscope, 134:2277–2281, 2024 [ABSTRACT FROM AUTHOR]

A neurological condition called dystonia results in abnormal, uncontrollable postures or movements because of sporadic or continuous muscular spasms. Several varieties of dystonia can impact people of all ages, leading to severe impairment and a decreased standard of living. The discovery of genes causing variations of single or mixed dystonia has improved our understanding of the disease's etiology. Genetic dystonias are linked to several genes, including pathogenic variations of VPS16, TOR1A, THAP1, GNAL, and ANO3. Diagnosis of dystonia is primarily based on clinical symptoms, which can be challenging due to overlapping symptoms with other neurological conditions, such as Parkinson's disease. This review aims to summarize recent advances in the genetic origins and management of focal dystonia. [ABSTRACT FROM AUTHOR]

Purpose: This study aims to aggregate and analyze existing clinical evidence to compare the efficacy and adverse effects of unilateral or bilateral botulinum toxin injections for the treatment of adductor spasmodic dysphonia (ADSD). Methods: Reports from non-randomized controlled trials and cohort studies pertaining to the efficacy and adverse effects of unilateral and bilateral botulinum toxin injections for ADSD were identified and retrieved from four electronic databases from inception to July 2023. The meta-analysis employed fixed or random effects models to assess pooled relative risks (RR), mean differences (MDs), and standard mean differences (SMDs) with their corresponding 95% confidence intervals (CIs). Results: We included two non-randomized controlled trials and seven cohort studies comprising 854 total patients. Meta-analysis of the included studies showed that bilateral botulinum toxin injections associated with a longer duration of vocal improvement (MD = − 2.89, 95% CI − 3.13 to − 2.65, I2 = 0%, P < 0.00001). However, bilateral botulinum toxin injections associated with an increase in adverse effects, including a longer duration of breathy voice quality (SMD = − 0.51, 95% CI − 0.79 to − 0.22, I2 = 35%, P = 0.0005) and a higher occurrence of swallowing difficulties (RR = 0.46, 95% CI 0.35 to 0.11, I2 = 0%, P < 0.00001). Conclusion: Bilateral botulinum toxin injections for ADSD showed a longer duration of vocal improvement, a longer breathy voice duration and a higher dysphagia occurrence and duration than unilateral injections. [ABSTRACT FROM AUTHOR]

Laryngeal dystonia is a debilitating disorder of voicing in which the laryngeal muscles are intermittently in spasm resulting in involuntary interruptions during speech. The central pathophysiology of laryngeal dystonia, underlying computational impairments in vocal motor control, remains poorly understood. Although prior imaging studies have found aberrant activity in the CNS during phonation in patients with laryngeal dystonia, it is not known at what timepoints during phonation these abnormalities emerge and what function may be impaired. To investigate this question, we recruited 22 adductor laryngeal dystonia patients (15 female, age range = 28.83-72.46 years) and 18 controls (eight female, age range = 27.40-71.34 years). We leveraged the fine temporal resolution of magnetoencephalography to monitor neural activity around glottal movement onset, subsequent voice onset and after the onset of pitch feedback perturbations. We examined event-related beta-band (12-30 Hz) and high-gamma-band (65-150 Hz) neural oscillations. Prior to glottal movement onset, we observed abnormal frontoparietal motor preparatory activity. After glottal movement onset, we observed abnormal activity in the somatosensory cortex persisting through voice onset. Prior to voice onset and continuing after, we also observed abnormal activity in the auditory cortex and the cerebellum. After pitch feedback perturbation onset, we observed no differences between controls and patients in their behavioural responses to the perturbation. But in patients, we did find abnormal activity in brain regions thought to be involved in the auditory feedback control of vocal pitch (premotor, motor, somatosensory and auditory cortices). Our study results confirm the abnormal processing of somatosensory feedback that has been seen in other studies. However, there were several remarkable findings in our study. First, patients have impaired vocal motor activity even before glottal movement onset, suggesting abnormal movement preparation. These results are significant because (i) they occur before movement onset, abnormalities in patients cannot be ascribed to deficits in vocal performance and (ii) they show that neural abnormalities in laryngeal dystonia are more than just abnormal responses to sensory feedback during phonation as has been hypothesized in some previous studies. Second, abnormal auditory cortical activity in patients begins even before voice onset, suggesting abnormalities in setting up auditory predictions before the arrival of auditory feedback at voice onset. Generally, activation abnormalities identified in key brain regions within the speech motor network around various phonation events not only provide temporal specificity to neuroimaging phenotypes in laryngeal dystonia but also may serve as potential therapeutic targets for neuromodulation.

BackgroundLaryngeal dystonia is a task-specific focal dystonia of laryngeal muscles that impairs speech and voice production. At present, there is no cure for LD. The most common therapeutic option for patients with LD involves Botulinum neurotoxin injections.ObjectiveProvide empirical evidence that non-invasive vibro-tactile stimulation (VTS) of the skin over the voice box can provide symptom relief to those affected by LD.MethodsSingle-group 11-week randomized controlled trial with a crossover between two dosages (20 min of VTS once or 3 times per week) self-administered in-home in two 4-week blocks. Acute effects of VTS on voice and speech were assessed in-lab at weeks 1, 6 and 11. Participants were randomized to receive either 40 Hz or 100 Hz VTS.Main outcome measuresPrimary: smoothed cepstral peak prominence (CPPS) of the voice signal to quantify voice and speech abnormalities, and perceived speech effort (PSE) ranked by participants as a measure of voice effort (scale 1–10). Secondary: number of voice breaks during continuous speech, the Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) inventory as a measure of overall disease severity and the Voice Handicap Index 30-item self report.ResultsThirty-nine people with a confirmed diagnosis of adductor-type LD (mean [SD] age, 60.3 [11.3] years; 18 women and 21 men) completed the study. A single application of VTS improved voice quality (median CPPS increase: 0.41 dB, 95% CI [0.20, 0.61]) and/or reduced voice effort (PSE) by at least 30% in up to 57% of participants across the three study visits. Effects lasted from less than 30 min to several days. There was no effect of dosage and no evidence that the acute therapeutic effects of VTS increased or decreased longitudinally over the 11-week study period. Both 100 and 40 Hz VTS induced measurable improvements in voice quality and speech effort. VTS induced an additional benefit to those receiving Botulinum toxin. Participants, not receiving Botulinum treatment also responded to VTS.ConclusionThis study provides the first systematic empirical evidence that the prolonged use of laryngeal VTS can induce repeatable acute improvements in voice quality and reductions of voice effort in LD.Clinical trial registrationClinicalTrials.gov ID: NCT03746509.

Objectives: Laryngeal dystonia (LD) is characterized by irregular and involuntary task-specific spasms of the intrinsic laryngeal muscles. There is no curative treatment for it, however, laryngeal botulinum neurotoxin injections (BoNT-I) are considered the standard of care therapy. This study aims to characterize the population of LD patients and to assess the results of laryngeal BoNT-I. Methods: A Retrospective cohort study was conducted. Medical records were reviewed for all the patients with LD diagnosis seen in the Voice Unit of the Red de Salud UC Christus between January 2013 and October 2021. Biodemographic, clinical and treatment data were collected. Additionally, a telephonic survey was completed by the patients that underwent laryngeal BoNT-I, including self-reported voice outcomes and Voice Handicap Index 10 (VHI-10). Results: Of the 34 patients with LD included in the study, 23 received a total of 93 laryngeal BoNT-I and 19 completed the telephone survey. The majority (97%) of the injections corresponded to patients with adductor LD and 3% to abductor LD. Patients received a median of 3 (1–17) injections, with a more frequent cricothyroid approach (94.4%), while the thyrohyoid approach accounted for 5.6% of cases. Most injections were bilateral (96.8%). A significant improvement in the vocal quality and effort was noted after the last injection and the overall BoNT-I treatment (P < 0.001). Similarly, the VHI-10 score improved from a median of 31 (7–40) to 2 (0–19) (P < 0.001) after the last injection. A post-treatment breathy voice was reported in 95% of patients, and dysphagia to liquids and solids in 68% and 21%, respectively. Conclusions: Laryngeal BoNT-I is an effective treatment for LD, achieving an improvement in self-reported vocal quality and VHI-10 scores, and a reduction of the self-reported vocal effort. Adverse effects are mild in the majority of cases, constituting a safe and effective therapy for these patients. [ABSTRACT FROM AUTHOR]

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Objectives: To evaluate spasmodic dysphonia patients' perception of pain associated with laryngeal botulinum toxin (BTX) injections and to determine factors associated with higher pain scores relative to other included patients. Methods: Prospective cohort study. Adult patients with adductor spasmodic dysphonia that presented to a tertiary laryngology practice for BTX injections were recruited from March to July 2022. Patients completed the visual analog scale (VAS) pre‐procedure to quantify predicted pain. Ten minutes post‐procedure they completed VAS and the short form McGill Pain Questionnaire (SF‐MPQ). Factors that may affect pain were extracted from charts. Descriptive statistics, univariate, and multivariate analyses were conducted (alpha = 0.05). Results: One hundred and nineteen patients were included (63 ± 14 yo, 26% Male). SF‐MPQ reported mild pain (4.12 ± 4.05 out of 45) with a pain intensity of none to mild (0.70 ± 0.89 out of 5). Bilateral injections yielded significantly higher SF‐MPQ scores (5.19 ± 4.66) than unilateral injections (3.30 ± 3.30) (p = 0.012). There was a significant VAS reduction from pre 28.9 ± 24.6 mm (out of 10 mm) to post 24.5 ± 22.3 (p < 0.001). On multiple regression analyses, receiving a bilateral injection significantly (p < 0.05) contributed to a model that predicted higher pre‐VAS (p = 0.013). Bilateral injections (p < 0.05) and higher VHI‐10 (p < 0.05) contributed to a model that predicted higher total SF‐MPQ (p = 0.001) and affective SF‐MPQ (p = 0.001) scores. Not being a professional voice user (PVU) significantly (p < 0.05) contributed to a model that predicted higher post‐VAS (p = 0.008) scores. Conclusions: BTX injections were well tolerated with low pain scores. Factors associated with higher relative predicted or experienced pain included bilateral versus unilateral injection, PVU status, and higher VHI‐10. Level of Evidence: 4 Laryngoscope, 134:240–246, 2024 [ABSTRACT FROM AUTHOR]

Abstract Objectives This study aims to analyze the impact of age and sex on botulinum neurotoxin (BoNT‐A) dosing and outcomes in adductor spasmodic dysphonia (AdSD). Methods A database review of all spasmodic dysphonia patients treated with BoNT from 1989 to 2018 at the Mayo Clinic in Arizona was performed. Only patients who had received ≥4 injections of BoNT‐A for AdSD were included. Patients were divided into two cohorts to analyze age, with an age of first treatment cutoff of 60 years. Patients were divided into male and female cohorts to analyze sex. Results The final analysis included 398 patients. The mean dose of BoNT‐A per treatment was significantly higher in the younger cohort (4.4 vs. 3.9 units, p = 0.048). The mean maximal benefit was similar (72% vs. 70%, p = 0.48); however, the mean length of benefit was significantly shorter in younger patients (3.0 vs. 3.6 months, p

Adductor spasmodic dysphonia (ADSD) is a neurological dystonia characterised by involuntary adductor spasms of the larynx during speech. Spasm frequency is often reported to increase during syllables that begin with voiced speech sounds, especially glottal stops. Because of its underlying physical and acoustic complexities, the voicing contrast in American English (AE) appears unlikely to interact consistently with a singular physical phenomenon like laryngeal spasm. This retrospective study investigated additional phonetic contrasts and their relationship to spasm frequency. Standardised, 144-word recordings of 36 participants with adductor spasmodic dysphonia were analysed. Productions were coded for rater-perceived syllable stress, voiced/voiceless onset, vowel/consonant onset, and word-onset place and manner of production. Phonetic contexts were compared using independent sample t-tests and Kruskal-Wallis statistics. Contexts in which spasm varied significantly included stressed/unstressed syllables, content/function words, and multisyllabic/monosyllabic words. Study results reaffirm the clinical usefulness of standardised ADSD/ABSD sentences during differential diagnosis but conflict with previous studies that report a connection between ADSD spasm and phoneme voicing. [ABSTRACT FROM AUTHOR]

Background/Objectives: Laryngeal sensory abnormality has been implicated as a component of adductor laryngeal dystonia (AdLD). The study objective was to assess laryngopharyngeal sensation in AdLD utilizing a calibrated, tactile aesthesiometer to deliver differential stimuli to lateral pyriform sinus (LPS), aryepiglottic fold (AEF), and false vocal fold (FVF). Methods: Patients with known Botox‐responsive AdLD underwent sensory testing using a previously‐validated methodology involving calibrated tactile stimuli (6–0, 5–0, 4.5–0, 4–0 nylon monofilaments). Laryngeal adductor reflex (LAR) and participant‐rated perceptual strength of stimulI were evaluated. Responses were compared to normative controls (n = 33). Two‐samples, Mann–Whitney and Fisher exact tests compared mean strength ratings and LAR between AdLD and control groups. Mixed‐effects logistic regression and linear models assessed association of filament size, stimulus site, age, sex, and LD status on LAR and perceptual strength rating respectively. Results: Thirteen AdLD patients (nine women, mean age 60+/−15 years) completed testing. Average LAR response rates were higher amongst all filament sizes in AdLD versus controls at LPS (56.3% vs. 35.7%) and AEF (96.1% vs. 70.2%) with comparable rates at FVF (90.2% vs. 91.7%). AdLD had 3.3 times the odds of observed LAR compared to controls (p = 0.005), but differences in subjective detection of stimuli, perceptual strength ratings, and cough/gag rates were insignificant on multivariate modeling (p > 0.05). Conclusions: This is the first study to objectively assess laryngopharyngeal sensation in AdLD. Findings demonstrated increased laryngopharyngeal sensation in AdLD compared to controls. The identification of increased laryngeal hypersensitivity in these patients may improve understanding of AdLD pathophysiology and identify future targets for intervention. Level of Evidence: 2 Laryngoscope, 133:2271–2278, 2023 [ABSTRACT FROM AUTHOR]

Objectives: The anterior, percutaneous Botulinum neurotoxin (BoNT) injection in the lateral cricoarytenoid muscle (LCA) guided by laryngeal electromyography (LEMG) is considered the golden standard treatment for several neurolaryngological disorders. The study presented in this article aims to assess the effectiveness of an alternative approach by which the injection is performed laterally under ultrasound monitoring. Study design: Anatomical dissection study in human cadavers. Settings: Academic health care center. Methods: Ultrasound-guided bilateral dye (0.1 mL of dye solution containing cold-curing polymers, latex, acrylates, acrylic esters, alcohol, and green color) injection in the LCA was performed by means of 24G needles and 1 mL syringes using the lateral approach. The dye location and distribution were assessed by anatomic dissection, performed immediately after the injection. Results: In 9/10 specimens, the dye was exclusively detectable in the LCA. In 1/10 case (left side), the dye could not be delivered in the LCA because of unintended penetration of the thyroid cartilage by the needle during injection. Anatomic dissection confirmed that the dye spread neither into the thyroarytenoid (TA) nor the cricothyroid muscle (CT). Conclusions: The anatomic dissection following lateral dye injection in the LCA under ultrasound guide confirmed the precision of this approach in delivery a substance exclusively in a pre-determined target. This feature makes this method an interesting addition or alternative to the standard LEMG-guided BoNT injection at least when the LCA is its target. Level of evidence: III. [ABSTRACT FROM AUTHOR]

Background: Despite many available patient‐reported outcome measures (PROMs) for laryngeal movement disorders, there is a lack of patient input regarding which PROM most accurately and conveniently captures aspects related to their vocal disease. This study aimed to assess patients' preferences among a selection of voice‐related PROMs (Voice Handicap Index‐10 [VHI‐10], OMNI‐Vocal Effort Scale [OMNI‐VES], Communicative Participation Item Bank‐General Short Form [CPIB‐10], and Visual Analog Scales [VAS]) within the laryngeal movement disorder population and investigate associations between selected instruments. Methods: Prior to botulinum toxin A injection, patients with laryngeal dystonia and/or essential tremor of the vocal tract were administered the VHI‐10, OMNI‐VES, CPIB‐10, and three novel VAS questions in a randomized order. Patients rank ordered the four PROMs based on the PROMs' reflection of their voice problems. Pearson's correlation coefficients evaluated pairwise associations among PROM scores. Fisher's exact test compared the preferred PROM rankings. Results: Seventy patients (53 female, mean age = 60.7 years) participated. The VHI‐10 and CPIB‐10 were most preferred at 33.9% and 27.4% respectively. The OMNI‐VES and VAS scales were less favored (19.4%, each). When analyzed by age ≥60 years, the CPIB‐10 was most favored (33.3%), but for age <60 years, VHI‐10 was most preferred (42.3%). There was a strong correlation between scores of all administered PROMs (strongest correlation between OMNI‐VES and VAS, r = 0.8, p < 0.001; the weakest correlation between OMNI‐VES and VHI‐10, r = 0.6, p < 0.001). Conclusions: With an increasing trend in PROMs usage and a strong correlation between all evaluated outcome instruments, insight regarding patients' PROM preferences is an area for further consideration. Level of Evidence: NA Laryngoscope, 133:1448–1454, 2023 For patients with laryngeal dystonia (formally called spasmodic dysphonia) and vocal tremors, there is no universally accepted outcome metric to assess and compare disease severity or response to treatment. Our study looks to determine which of the currently used tools/questionnaires are most preferred by patients themselves. [ABSTRACT FROM AUTHOR]

Purpose: The purpose of this study was to determine whether patient voicerelated diagnosis, severity of dysphonia, and rater’s experience influence the relationship between laryngeal oscillation ratings made from videostroboscopic and high-speed videoendoscopic (HSV) exams. Method: Stroboscopy and HSV exams from 15 patients with adductor spasmodic dysphonia (ADSD) and 15 with benign vocal fold lesions were rated for laryngeal oscillation and closure by 10 licensed speech-language pathologists (SLPs). Raters were divided into low- (< 5 years) and high-experience (> 5 years) groups. Ratings of vocal fold amplitude, mucosal wave, periodicity, phase symmetry, nonvibrating portion of the vocal fold, and glottal closure were examined using an online form adapted from the Voice Vibratory Assessment of Laryngeal Imaging (VALI). Results: Stroboscopy and HSV ratings were more strongly positively correlated for patients with benign vocal fold lesions (r between .43 and .75) than for those with ADSD (r between .40 and .68). Differences between stroboscopy and HSV exams were significantly greater for ratings of amplitude, mucosal wave, and periodicity in patients with ADSD than for patients with benign vocal fold lesions. Raters with < 5 years of experience showed significantly greater differences between stroboscopy and HSV ratings of amplitude and nonvibrating portion of the vocal fold for patients with ADSD only. Significantly greater differences between ratings of periodicity and phase symmetry were observed in patients with more severe dysphonia. Conclusions: Differences in laryngeal ratings made between HSV and stroboscopy exams may be influenced by patient diagnosis, severity of dysphonia, and rater experience. Future study is warranted to determine how the differences observed influence clinical diagnosis and outcomes. [ABSTRACT FROM AUTHOR]

The cortical silent period (cSP) is a period of electrical silence following a motor-evoked potential (MEP) in the electromyographic signal recorded from a muscle. The MEP can be elicited by transcranial magnetic stimulation (TMS) over the primary motor cortex site corresponding with the muscle. The cSP reflects the intracortical inhibitory process mediated by GABAA and GABAB receptors. The study aimed to investigate the cSP in the cricothyroid (CT) muscle after applying e-field-navigated TMS over the laryngeal motor cortex (LMC) in healthy subjects. Then, a cSP as a neurophysiologic feature for laryngeal dystonia was observed. We applied a single-pulse e-field-navigated TMS to the LMC over both hemispheres with hook-wire electrodes positioned in the CT muscle in nineteen healthy participants, which triggered the elicitation of contralateral and ipsilateral corticobulbar MEPs. The subjects were engaged in a vocalization task, and then we assessed the following metrics: LMC intensity, peak-to-peak MEP amplitude in the CT muscle, and cSP duration. The results showed that the cSP duration from the contralateral CT muscle was distributed from 40 ms to 60.83 ms, and from the ipsilateral CT muscle, from 40 ms to 65.58 ms. Also, no significant difference was found between the contralateral and ipsilateral cSP duration (t(30) = 0.85, p = 0.40), MEP amplitude in the CT muscle (t(30) = 0.91, p = 0.36), and LMC intensity (t(30) = 1.20, p = 0.23). To conclude, the applied research protocol showed the feasibility of recording LMC corticobulbar MEPs and observing the cSP during vocalization in healthy participants. Furthermore, an understanding of neurophysiologic cSP features can be used to study the pathophysiology of neurological disorders that affect laryngeal muscles, such as laryngeal dystonia. [ABSTRACT FROM AUTHOR]

Adductor spasmodic dysphonia is a type of adult-onset focal dystonia characterized by involuntary spasms of laryngeal muscles. This paper applied machine learning techniques for the severity assessment of spasmodic dysphonia. To this aim, 7 perceptual indices and 48 acoustical parameters were estimated from the Italian word /a'jwɔle/ emitted by 28 female patients, manually segmented from a standardized sentence and used as features in two classification experiments. Subjects were divided into three severity classes (mild, moderate, severe) on the basis of the G (grade) score of the GRB scale. The first aim was that of finding relationships between perceptual and objective measures with the Local Interpretable Model-Agnostic Explanations method. Then, the development of a diagnostic tool for adductor spasmodic dysphonia severity assessment was investigated. Reliable relationships between G; R (Roughness); B (Breathiness); Spasmodicity; and the acoustical parameters: voiced percentage, F2 median, and F1 median were found. After data scaling, Bayesian hyperparameter optimization, and leave-one-out cross-validation, a k-nearest neighbors model provided 89% accuracy in distinguishing patients among the three severity classes. The proposed methods highlighted the best acoustical parameters that could be used jointly with GRB indices to support the perceptual evaluation of spasmodic dysphonia and provide a tool to help severity assessment of spasmodic dysphonia. [ABSTRACT FROM AUTHOR]

Spasmodic dysphonia (SD) is considered a rare focal laryngeal dystonia characterized by task-specific voice dysfluency resulting from selective intrinsic laryngeal musculature hyperfunction. Symptoms may be attenuated by a sensory trick. Although SD can be seen at times in generalized dystonia syndrome, it is typically a sporadic phenomenon, and the involvement of the laryngeal adductor muscles is more common than that of the abductor muscles. This research reviews the literature for the pathogenesis, clinical characteristics, treatment options, and current management methods of SD. Technological advances have enabled clinicians to better understand the connection between laryngeal function and dysfunction. Refinements in imaging and genetic investigation techniques have helped better understand the underlying mechanisms of this neurolaryngology disorder. Currently, the standard of care for SD is the symptomatic management of botulinum toxin (BT) chemodenervation. This is supported by a large body of literature attesting to its efficacy in many different research studies, particularly in the uncomplicated adductor form of the disorder. Efforts towards surgical treatment predate the development of BT treatment by a decade, but the long-term efficacy has not been proven and, further research is expected. Symptom relief in patients with abductor SD and dystonia with tremors after surgical and BT treatments and those in patients remains suboptimal. [ABSTRACT FROM AUTHOR]

Laryngeal dystonia (LD), or spasmodic dysphonia (SD), is a chronic, task-specific, focal movement disorder affecting the larynx. It interferes primarily with the essential functions of phonation and speech. LD affects patients' ability to communicate effectively and significantly diminishes their quality of life. Botulinum neurotoxin was first used as a therapeutic agent in the treatment of LD four decades ago and remains the standard of care for the treatment of LD. This article provides an overview of the clinical application of botulinum neurotoxin in the management of LD, focusing on the classification for this disorder, its pathophysiology, clinical assessment and diagnosis, the role of laryngeal electromyography and a summary of therapeutic injection techniques, including a comprehensive description of various procedural approaches, recommendations for injection sites and dosage considerations. [ABSTRACT FROM AUTHOR]

The prevalence of dystonia has been studied since the 1980s. Due to different methodologies and due to varying degrees of awareness, resulting figures have been extremely different. We wanted to determine the prevalence of dystonia according to its current definition, using quality-approved registries and based on its relevance for patients, their therapy and the health care system. We applied a service-based chart review design with the City of Hannover as reference area and a population of 525,731. Barrier-free comprehensive dystonia treatment in few highly specialised centres for the last 30 years should have generated maximal dystonia awareness, a minimum of unreported cases and a high degree of data homogeneity. Prevalence [n/1mio] and relative frequency is 601.1 (100%) for all forms of dystonia, 251.1 (42%) for cervical dystonia, 87.5 (15%) for blepharospasm, 55.2 (9%) for writer's cramp, 38.0 (6%) for tardive dystonia, 32.3 (5%) for musician's dystonia, 28.5 (5%) for psychogenic dystonia, 26.6 (4%) for generalised dystonia, 24.7 (4%) for spasmodic dysphonia, 20.9 (3%) for segmental dystonia, 15.2 (3%) for arm dystonia and 13.3 (2%) for oromandibular dystonia. Leg dystonia, hemidystonia and complex regional pain syndrome-associated dystonia are very rare. Compared to previous meta-analytical data, primary or isolated dystonia is 3.3 times more frequent in our study. When all forms of dystonia including psychogenic, generalised, tardive and other symptomatic dystonias are considered, our dystonia prevalence is 3.7 times higher than believed before. The real prevalence is likely to be even higher. Having based our study on treatment necessity, our data will allow better allocation of resources for comprehensive dystonia treatment. [ABSTRACT FROM AUTHOR]

Purpose: Traditional vocal fold pathology recognition typically requires expertise of laryngologists and advanced instruments, primarily through direct visualization. This study aims to augment this conventional paradigm by introducing a parallel diagnostic procedure. Our objective is to harness a machine-learning algorithm designed to discern intricate patterns within patients' voice recordings to distinguish not only between healthy and hoarse voices but also among various specific disorders., Materials and Methods: We employed a machine-learning algorithm, utilizing transfer learning on the HuBERT model with Saarbruecken Voice Database samples. The study was conducted in two stages: a binary classifier distinguishes healthy and hoarse voices, while a subsequent multi-class classifier identifies specific voice disorders. Data from 2103 sessions, including over 25,000 components, representing diverse pathologies and healthy individuals, was analyzed. The models were trained, validated, and tested with a focus on robustness and accuracy in diagnosis., Results: The binary classifier achieved 82 % accuracy in distinguishing healthy from pathological voices. The multi-class algorithm which aims to identify specific laryngeal disorders obtained the highest accuracy (>93 %) for Laryngeal Dystonia. Noteworthy is the persistent challenge posed by Laryngeal Dystonia, a condition lacking a definitive diagnostic modality., Conclusions: Our findings demonstrate the feasibility of utilizing machine-learning algorithms to process voice samples, categorizing them into distinct pathologies. This approach holds potential for enhance patient triage, streamline diagnostics, and elevate overall patient care. Particularly valuable for challenging diagnoses, such as Laryngeal Dystonia, this method underscores the transformative role of machine learning in optimizing healthcare practices., Competing Interests: Declaration of competing interest This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors., (Copyright © 2024 The Authors. Published by Elsevier Inc. All rights reserved.)

A 45-year-old woman with recurrent dyspnea for 40 years was previously diagnosed with bronchial asthma and spasmodic dysphonia. On admission, the patient was diagnosed with expiratory central airway collapse (ECAC) due to expiratory dynamic airway collapse based on radiographic examination, chest computed tomography, and bronchoscopy. After continuous positive airway pressure and temporal airway stenting, surgical tracheobronchoplasty and tracheal membranous portion reinforcement using polypropylene mesh successfully relieved the respiratory symptoms. In patients with airway obstructive disease refractory to conventional therapies, ECAC should be considered.

Objective: Currently, no clinically feasible objective measures exist that are specific to the signs of adductor laryngeal dystonia (LD), deterring effective diagnosis and treatment. This project sought to establish concurrent validity of a new automated acoustic outcome measure, designed to be specific to adductor laryngeal dystonia (AdLD): the spectral aggregate of the high-passed fundamental frequency contour (SAHfo). Method: Twenty speakers with AdLD read voiced phoneme-loaded (more symptomatic) and voiceless phoneme-loaded (less symptomatic) sentences. LD discontinuities (defined as phonatory breaks, frequency shifts, and creak), the acoustic ramifications of laryngeal spasms, were manually identified. The frequency content of the fo contour was examined as a function of time, and content above 1000 Hz was summed to automatically calculate SAHfo. Multiple linear regression analysis was applied to SAHfo based on LD discontinuities and sentence type (voiced or voiceless phoneme-loaded). Results: The regression model accounted for 41.1% of the variance in SAHfo. Both the LD discontinuities and sentence type were statistically related to SAHfo. Conclusion: Results of this study provide evidence of concurrent validity. SAHfo is an automatic outcome measure specific to acoustic signs of AdLD that may be useful to track treatment progress. [ABSTRACT FROM AUTHOR]

Objective: Adductor spasmodic dysphonia (AdSD) is caused by hyperadduction of the vocal folds during phonation, resulting in a strained voice. Animal models are not yet used to elucidate this intractable disease because AdSD has a difficult pathology without a definitive origin. For the first step, we established an animal model with vocal fold hyperadduction and evaluated its validity by assessing laryngeal function.Methods: In this experimental animal study, three adult Japanese 20-week-old rabbits were used. The models were created using a combination of cricothyroid approximation, forced airflow, and electrical stimulation of the recurrent laryngeal nerves (RLNs). Cricothyroid approximation was added to produce a glottal slit. Thereafter, both RLNs were electrically stimulated to induce vocal fold hyperadduction. Finally, the left RLN was transected to relieve hyperadduction. The sound, endoscopic images, and subglottal pressure were recorded, and acoustic analysis was performed.Results: Subglottal pressure increased significantly, and the strained sound was produced after the electrical stimulation of the RLNs. After transecting the left RLN, the subglottal pressure decreased significantly, and the strained sound decreased. Acoustic analysis revealed an elevation of the standard deviation of F0 (SDF0) and degree of voice breaks (DVB) through stimulation of the RLNs, and degradation of SDF0 and DVB through RLN transection. Formant bands in the sound spectrogram were interrupted by the stimulation and appeared again after the RLN section.Conclusion: This study developed a rabbit model with vocal fold hyperadduction . The subglottal pressure and acoustic analysis of this model resembled the characteristics of patients with AdSD. This model could be helpful to elucidate the pathology of the larynx caused by hyperadduction, and evaluate and compare the treatments for strained phonation. [ABSTRACT FROM AUTHOR]

Purpose: This study examined the relationship between voice quality and glottal geometry dynamics in patients with adductor spasmodic dysphonia (ADSD). Method: An objective computer vision and machine learning system was developed to extract glottal geometry dynamics from nasolaryngoscopic video recordings for 78 patients with ADSD. General regression models were used to examine the relationship between overall voice quality and 15 variables that capture glottal geometry dynamics derived from the computer vision system. Two experts in ADSD independently rated voice quality for two separate voice tasks for every patient, yielding four different voice quality rating models. Results: All four of the regression models exhibited positive correlations with clinical assessments of voice quality (R2s = .30-.34, Spearman rho = .55-.61, all with p < .001). Seven to 10 variables were included in each model. There was high overlap in the variables included between the four models, and the sign of the correlation with voice quality was consistent for each variable across all four regression models. Conclusion: We found specific glottal geometry dynamics that correspond to voice quality in ADSD. [ABSTRACT FROM AUTHOR]

Purpose: Spasmodic dysphonia (SD) or laryngeal dystonia is as a rare vocal disorder characterized by involuntary action-induced endolaryngeal contraction. In the last decade, botulin toxin injection has become the standard treatment in adductor spasmodic dysphonia necessitating repetitive injections. The purpose of this study is to analyze retrospectively data from patients treated with the minimal-invasive transoral radiofrequency-induced thermotherapy (RFITT) of the terminal branches of the recurrent nerve. Methods: Between 2009 and 2015, 11 patients (six females and five males aged from 32 to 91 years) with adductor SD were treated with RFITT. Pre-operative and post-operative vocal assessments (VHI-30, GRBASI, and acoustic-aerodynamics measurements), number of surgical revisions, delay between procedures, and post-operative complications were recorded. Statistical analyses were carried out on the first vocal assessment performed 2–8 weeks after the first procedure. Results: Based on available data from ten patients, voice handicap index (VHI) showed improvement with a mean value of −17.7 points (p-value (pval) = 0.014, adjusted p-value (adj pval) = 0.21); instability has also revealed improvement in six patients (pval = 0.05, adj pval = 0.31). Four patients underwent only one procedure including one patient showing still long-term beneficial results after 5 years of follow-up. Other patients required one to three new procedures with an average time between procedures of 15.3 months. Over 24 surgeries performed on a total of 11 patients, one definitive treatment-related severe adverse event was reported. Conclusion: Thanks to long-lasting effect, repetitive treatments are less frequent compared to botulin toxin therapy. In our opinion, RFITT is a promising alternative to botulin toxin as a second-step procedure in case of toxin resistance or patient's lack of compliance. [ABSTRACT FROM AUTHOR]

Keywords: spasmodic dysphonia; ATX-AFG3L2; SCA28; AFG3L2; ataxia EN spasmodic dysphonia ATX-AFG3L2 SCA28 AFG3L2 ataxia 1024 1026 3 06/20/23 20230601 NES 230601 Heterozygous pathogenic variants in ATPase family gene 3-like 2 ( I AFG3L2 i ) gene are associated with autosomal dominant cerebellar ataxia (ATX- I AFG3L2 i ), previously known as spinocerebellar ataxia type 28, which typically presents with slowly progressive cerebellar ataxia, dysarthria, oculomotor abnormalities, and pyramidal signs.[[1]] Carriers of compound heterozygous I AFG3L2 i variants may demonstrate other features including ptosis, optic atrophy, external ophthalmoplegia, generalized dystonia, and lingual dyskinesia.[[3], [5]] Here, we report an individual with a rare compound heterozygous I AFG3L2 i variant who uniquely presented with spasmodic dysphonia, along with other typical features of ATX- I AFG3L2 i . Spasmodic Dysphonia in a Patient with Spinocerebellar Ataxia Associated with a Rare AFG3L2 Variant (ATX-AFG3L2) ATX-AFG3L2, spasmodic dysphonia, AFG3L2, SCA28, ataxia. [Extracted from the article]