Exploring the Neural Bases of Primary Muscle Tension Dysphonia: A Case Study Using Functional Magnetic Resonance Imaging.

Primary muscle tension dysphonia (pMTD) is a voice disorder that occurs in the absence of laryngeal pathology. Dysregulated activity of the paralaryngeal muscles is considered the proximal cause; however, the central origin of this aberrant laryngeal muscle activation is unclear. The Trait Theory (Roy and Bless, 2000a,b) proposed that specific personality traits can predispose one to laryngeal motor inhibition and pMTD, and this inhibition is mediated by a hyperactive "behavioral inhibition system (BIS)" composed of limbic system structures (and associated prefrontal connections). This case study used functional magnetic resonance imaging to detect brain activation changes associated with successful management of pMTD, thereby evaluating possible neural correlates of this poorly understood disorder. METHOD: A 61-year-old woman with moderate-to-severe pMTD underwent functional magnetic resonance imaging scans before and immediately after successful treatment using manual circumlaryngeal techniques. Experimental stimuli were blocks of repeated vowel production and overt sentence reading. RESULTS: Significantly greater activation was observed pre- versus posttreatment in all regions of interest during sentence production, that is, periaqueductal gray, amygdala, hypothalamus, anterior cingulate cortex, hippocampus, dorsolateral prefrontal cortex, Brodmann area 10, and premotor and inferior sensorimotor cortex. CONCLUSIONS: Our findings are compatible with overactivation of neural regions associated with the BIS (cingulate cortex, amygdala, hypothalamus, periaqueductal gray) and motor inhibition networks (eg, [pre-]supplementary motor area) along with the dorsolateral prefrontal cortex and medial prefrontal cortex. Heightened input from limbic regions combined with dysfunctional prefrontal regulation may interfere with laryngeal motor preparation, initiation, and execution thereby contributing to disordered voice in pMTD.

Manual laryngeal reposturing as a primary approach for mutational falsetto.

OBJECTIVES/HYPOTHESIS: Mutational falsetto is a functional voice disorder characterized by failure of the male high-pitched preadolescent voice to transition to the lower pitch of adolescence and adulthood. Few objective outcomes data exist regarding the effectiveness of voice therapy for this poorly understood disorder. This study examined the immediate effects of a single therapy session using manual laryngeal reposturing as a primary approach in the management of mutational falsetto. STUDY DESIGN: Retrospective case series, pre-/posttreatment. METHODS: Manual circumlaryngeal techniques, including digital laryngeal reposturing maneuvers, were used as the primary approach to treat 12 consecutive males with mutational falsetto (mean age 17.5 yrs., range 14-25 years). Pre- and posttreatment audiorecordings of connected speech and sustained vowel samples were submitted to auditory-perceptual and acoustic analysis to assess the effects of a single voice therapy session. Acoustic estimates of severity in both connected speech and sustained vowel productions were computed using the Cepstral Spectral Index of Dysphonia (CSID), a multivariate dysphonia summary tool that incorporates cepstral and spectral measures. RESULTS: Pre- and posttreatment comparisons confirmed a significant change in the direction of normal vocal function within a single treatment session for both listener- and CSID-based estimates of dysphonia severity. CONCLUSION: Behavioral therapy using manual laryngeal reposturing by an experienced voice clinician is an effective and efficient primary approach for mutational falsetto. LEVEL OF EVIDENCE: 4. Laryngoscope, 127:645-650, 2017.

Objective Measures of Swallowing Function Applied to the Dysphagia Population: A One Year Experience.

Quantitative, reliable measures of swallowing physiology can be made from an modified barium swallowing study. These quantitative measures have not been previously employed to study large dysphagic patient populations. The present retrospective study of 139 consecutive patients with dysphagia seen in a university tertiary voice and swallowing clinic sought to use objective measures of swallowing physiology to (1) quantify the most prevalent deficits seen in the patient population, (2) identify commonly associated diagnoses and describe the most prevalent swallowing deficits, and (3) determine any correlation between objective deficits and Eating Assessment Tool (EAT-10) scores and body mass index. Poor pharyngeal constriction (34.5 %) and airway protection deficits (65.5 %) were the most common swallowing abnormalities. Reflux-related dysphagia (36 %), nonspecific pharyngeal dysphagia (24 %), Parkinson disease (16 %), esophageal abnormality (13 %), and brain insult (10 %) were the most common diagnoses. Poor pharyngeal constriction was significantly associated with an esophageal motility abnormality (p < 0.001) and central neurologic insult. In general, dysphagia symptoms as determined by the EAT-10 did not correlate with swallowing function abnormalities. This preliminary study indicates that reflux disease is common in patients with dysphagia and that associated esophageal abnormalities are common in dysphagic populations and may be associated with specific pharyngeal swallowing abnormalities. However, symptom scores from the EAT-10 did not correspond to swallowing pathophysiology.

Outcomes of Laryngeal Reinnervation for Unilateral Vocal Fold Paralysis in Children: Associations With Age and Time Since Injury.

OBJECTIVE: Outcomes of laryngeal reinnervation with ansa-cervicalis for unilateral vocal fold paralysis (UVFP) may be influenced by age of the patient and time interval between laryngeal nerve injury and reinnervation, suggesting less favorable outcomes in older patients and greater than 2-year time interval after injury. This study examines these issues in the pediatric population. METHOD: Review of prospectively collected data set of 35 children and adolescents (1-21 years) that underwent ansa-recurrent laryngeal nerve (RLN) laryngeal reinnervation for UVFP. RESULTS: The time from RLN injury to reinnervation averaged 5.0 years (range, 0.8-15.2 years). No correlation was found between age at reinnervation (r = 0.15) and patient- or parent-reported global percentage voice outcome or perceptual ratings. There was slight negative correlation in duration between RLN injury and reinnervation and voice outcomes (r = -0.31). Postoperative voice self/surrogate global percentage rating average was 80.5% (range, 50%-100%), and perceptual rating GRBAS sum score average was 2.9 (range, 0-7). CONCLUSION: In pediatric ansa-RLN reinnervation for UVFP, no correlation between age at surgery and postoperative outcome was found. Denervation duration showed slight negative correlation, similar to what has been reported in adults, though voice improvement was seen in all patients.

Quantitative Measures of Swallowing Deficits in Patients With Parkinson's Disease.

OBJECTIVE: Dysphagia and associated aspiration pneumonia are commonly reported sequelae of Parkinson's disease (PD). Previous studies of swallowing in patients with PD have described prolonged pharyngeal transit time, delayed onset of pharyngeal transit, cricopharyngeal (CP) achalasia, reduced pharyngeal constriction, and slowed hyolaryngeal elevation. These studies were completed using inconsistent evaluation methodology, reliance on qualitative analysis, and a lack of a large control group, resulting in concerns regarding diagnostic precision. The purpose of this study was to investigate swallowing function in patients with PD using a norm-referenced, quantitative approach. METHODS: This retrospective study includes 34 patients with a diagnosis of PD referred to a multidisciplinary voice and swallowing clinic. Modified barium swallow studies were performed using quantitative measures of pharyngeal transit time, hyoid displacement, CP sphincter opening, area of the pharynx at maximal constriction, and timing of laryngeal vestibule closure relative to bolus arrival at the CP sphincter. RESULTS: Reduced pharyngeal constriction was found in 30.4%, and a delay in airway closure relative to arrival of the bolus at the CP sphincter was the most common abnormality, present in 62% of patients. Previously reported findings of prolonged pharyngeal transit, poor hyoid elevation, and CP achalasia were not identified as prominent features.

Voice, speech, and laryngeal features of primary Sjögren's syndrome.

OBJECTIVE: This study examined voice, speech, and laryngeal characteristics in primary Sjögren's syndrome (pSS). METHODS: Eleven patients (10 female, 1 male; mean [SD] age = 57 [14] years) from The University of Utah Division of Rheumatology provided connected speech and sustained vowel samples. Analyses included the Multi-Dimensional Voice Profile, the Analysis of Dysphonia in Speech and Voice, and dysphonia severity, speech clarity, and videolaryngostroboscopy ratings. RESULTS: Shimmer, amplitude perturbation quotient, and average fundamental frequency differed significantly from normative values (P < .01). Cepstral Spectral Index of Dysphonia values indicated mild-to-moderate dysphonia in connected speech (mean [SD] = 20.26 [8.36]) and sustained vowels (mean [SD] = 16.91 [11.08]). Ratings of dysphonia severity and speech clarity using 10-cm visual analog scales suggested mild-to-moderate dysphonia in connected speech (mean [SD] = 2.11 [1.72]) and sustained vowels (mean [SD] = 3.13 [2.20]) and mildly reduced speech clarity (mean [SD] = 1.46 [1.36]). Videolaryngostroboscopic ratings indicated mild-to-moderate dryness and mild reductions in overall laryngeal function. Voice Handicap Index scores indicated mild-to-moderate voice symptoms (mean [SD] = 43 [23]). CONCLUSION: Individuals with pSS may experience dysphonia and articulatory imprecision, typically in the mild-to-moderate range. These findings have implications for diagnostic and referral practices in pSS.

Comparing nebulized water versus saline after laryngeal desiccation challenge in Sjögren's Syndrome.

OBJECTIVES/HYPOTHESIS: This study examined the effects of a laryngeal desiccation challenge and two nebulized hydration treatments on phonation threshold pressure (PTP), vocal effort, and throat dryness in patients with chronic airway dryness. STUDY DESIGN: Double-blind, within-subjects crossover design. METHODS: Eleven individuals with Primary Sjögren's Syndrome received a 15-minute laryngeal desiccation challenge (breathing dry air-<1% relative humidity-transorally), followed by nebulized isotonic saline or nebulized water treatments (3 mL) on 2 consecutive weeks. PTP, as well as self-perceived vocal effort, mouth, and throat dryness were assessed before and after the desiccation challenge, and at 5, 35, and 65 minutes after the nebulized treatment. RESULTS: The laryngeal desiccation challenge produced statistically significant increases in PTP, vocal effort, and mouth and throat dryness (P < 0.05). Nebulized saline produced greater-but not statistically significant-treatment effects than water. PTP was more correlated with throat dryness than vocal effort. CONCLUSION: Patients with chronic airway dryness experienced phonatory changes following dry air exposure. Nebulized isotonic saline may offset this effect. Future research should explore dose-response relationships among dry air exposure, nebulized treatments, voice change, and self-perceived throat dryness.

Case-control study of risk factors for spasmodic dysphonia: A comparison with other voice disorders.

OBJECTIVES/HYPOTHESIS: This epidemiology study examined risk factors uniquely associated with spasmodic dysphonia (SD). STUDY DESIGN: Case-control. METHODS: A questionnaire was administered to 150 patients with SD (with and without coexisting vocal tremor) and 136 patients with other structural, neurological, and functional voice disorders (excluding SD and vocal tremor). Questions included personal and family medical histories, environmental exposures, trauma, illnesses, voice use habits, and the Short Form 36. RESULTS: Several factors were uniquely associated with SD (α = .05), including: 1) a personal history of cervical dystonia, sinus and throat illnesses, mumps, rubella, dust exposure, and frequent volunteer voice use, 2) a family history of voice disorders, 3) an immediate family history of vocal tremor and meningitis, and 4) an extended family history of head and neck tremor, ocular disease, and meningitis. Vocal tremor coexisted with SD in 29% of cases. Measles and mumps vaccines were protective for SD. CONCLUSIONS: SD is likely multifactorial and associated with several endogenous and exogenous factors. Certain viral exposures, voice use patterns, and familial neurological conditions may contribute to the onset of SD later in life.

Spasmodic dysphonia: onset, course, socioemotional effects, and treatment response.

OBJECTIVES: This investigation explored the onset, progression, socioemotional effects, and treatment outcomes of spasmodic dysphonia (SD). METHODS: A cross-sectional epidemiological approach was used to examine questionnaire responses from 150 individuals with SD. RESULTS: Symptoms of SD (mean age at onset, 46 years) began gradually in 76% of cases and were progressive (ie, failed to plateau) in 34% of cases. Botulinum toxin A (Botox) helped to attenuate voice symptoms in 91% of cases; however, the scores on the Voice-Related Quality of Life questionnaire (V-RQOL) were not associated with this effect. The V-RQOL scores improved with time since symptom onset, independent of age and treatment. The patients with only SD experienced onset, course, and progression of symptoms similar to those of the patients with SD and coexisting vocal tremor. CONCLUSIONS: The symptoms of SD begin gradually and worsen over time. New evidence indicates that SD symptoms may continue to progress without plateau in at least a subset of patients. Individuals with SD and coexisting vocal tremor experience symptom trajectories similar to those of patients with SD only. Although Botox may attenuate voice symptoms, these effects do not appear to be strongly related to the V-RQOL scores. These results provide new and valuable insights regarding the onset, course, progression, and treatment of SD.

Laryngeal features of external superior laryngeal nerve denervation: revisiting a century-old controversy.

A long-standing controversy exists regarding the laryngoscopic features associated with unilateral denervation of the external superior laryngeal nerve (ESLN). Recently, we modeled acute unilateral cricothyroid muscle paralysis by blocking the ipsilateral ESLN with lidocaine hydrochloride, and identified epiglottic petiole deviation to the side of paralysis during high-pitched voice production as a possible diagnostic sign. This study provides preliminary clinical evidence supporting the presence of petiole deviation in cases of ESLN denervation. Epiglottic petiole deviation to the side of weakness was present in electromyographically confirmed cases of unilateral partial or complete ESLN denervation, in isolation or in combination with denervation of other branches of the vagus nerve. In addition, a case of complete ESLN and recurrent laryngeal nerve (RLN) denervation showed return of the petiole to the midline 6 months after surgical reinnervation of the ESLN and RLN. Finally, petiole deviation was not present in isolated RLN paralysis--a finding suggesting that the diagnostic sign is uniquely associated with ESLN denervation. We concluded that deviation of the petiole to the side of cricothyroid muscle weakness during high-pitched voice production represents a potential diagnostic sign of unilateral ESLN denervation. Further research is necessary to determine factors that influence the expression and detection of this sign, as well as its diagnostic precision.

Risk and protective factors for spasmodic dysphonia: a case-control investigation.

OBJECTIVES: Spasmodic dysphonia (SD) is a chronic, incurable, and often disabling voice disorder of unknown pathogenesis. The purpose of this study was to identify possible endogenous and exogenous risk and protective factors uniquely associated with SD. STUDY DESIGN: Prospective, exploratory, case-control investigation. METHODS: One hundred fifty patients with SD and 150 medical controls (MCs) were interviewed regarding their personal and family histories, environmental exposures, illnesses, injuries, voice use patterns, and general health using a previously vetted and validated epidemiologic questionnaire. RESULTS: Odds ratios and multiple logistic regression analyses (α<0.15) identified several factors that significantly increased the likelihood of having SD. These factors included (1) a personal history of mumps, blepharospasm, tremor, intense occupational and avocational voice use, and a family history of voice disorders; (2) an immediate family history of meningitis, tremor, tics, cancer, and compulsive behaviors; and (3) an extended family history of tremor and cancer. CONCLUSIONS: SD is likely multifactorial in etiology, involving both genetic and environmental factors. Viral infections/exposures, along with intense voice use, may trigger the onset of SD in genetically predisposed individuals. Future studies should examine the interaction among genetic and environmental factors to determine the pathogenesis of SD.

Vocal function exercises for presbylaryngis: a multidimensional assessment of treatment outcomes.

OBJECTIVES: Presbylaryngis, or aging of the larynx, can adversely affect vocal function and quality of life in the elderly. This preliminary investigation examined the effects of vocal function exercises, a physiologic voice therapy approach, as a primary treatment for presbylaryngis. METHODS: Nine consecutive elderly patients with presbylaryngis (2 female, 7 male) underwent a 6-week course of voice therapy employing vocal function exercises. Pretherapy-versus-posttherapy comparisons were made of self-ratings of voice handicap and phonatory effort level, as well as auditory-perceptual voice assessments, acoustic analyses, and visual-perceptual evaluations of laryngeal images. RESULTS: After treatment, patients reported significant reductions on Voice Handicap Index scores, phonatory effort levels, and voice disorder severity. Blinded listeners rated the posttreatment voices as significantly less breathy and strained. However, comparison of pretreatment and posttreatment maximum phonation times, acoustic measures, and laryngeal images did not reveal significant changes. CONCLUSIONS: These preliminary data suggest that vocal function exercises produce significant functional and perceptual improvements in voice, and deserve further attention as a treatment for elderly patients with presbylaryngis.

Nebulized isotonic saline versus water following a laryngeal desiccation challenge in classically trained sopranos.

PURPOSE: To examine the effects of nebulized isotonic saline (IS) versus sterile water (SW) on self-perceived phonatory effort (PPE) and phonation threshold pressure (PTP) following a surface laryngeal dehydration challenge in classically trained sopranos. METHOD: In a double-blind, within-subject crossover design, 34 sopranos breathed dry air (relative humidity < 1%) transorally for 15 min and then nebulized 3 mL of IS or SW, or experienced a no-treatment control condition over 3 consecutive weeks. PPE and PTP were measured every 15 min from baseline through 2 hr postdesiccation. RESULTS: PPE increased significantly following the laryngeal desiccation challenge in all 3 treatment conditions (p < .01). After nebulization, PPE returned to baseline for the IS condition only. For the SW and control conditions, PPE remained above baseline during the 2 hr after desiccation. No statistically significant changes in PTP following laryngeal desiccation were observed, although values for the IS condition remained below baseline for nearly 2 hr after nebulization. PPE and PTP were not significantly correlated. CONCLUSIONS: Following a laryngeal surface dehydration challenge, classically trained sopranos reported increased vocal effort that persisted for at least 2 hr. Compared with SW, nebulized IS showed promise as an effective way to remediate the adverse, self-perceived effects of laryngeal desiccation.

Differential diagnosis of muscle tension dysphonia and adductor spasmodic dysphonia using spectral moments of the long-term average spectrum.

OBJECTIVES/HYPOTHESIS: Adductor spasmodic dysphonia (ADSD) can mimic the voice characteristics of muscle tension dysphonia (MTD) contributing to diagnostic confusion and inappropriate management. Elevated spectral noise has been reported in MTD, which may aid in differential diagnosis. The long-term average spectrum (LTAS) can be compared to a Gaussian bell curve using spectral moments analysis. Four moments describe features of the LTAS: spectral mean (moment 1), standard deviation (moment 2), skewness (moment 3), and kurtosis (moment 4). This investigation evaluated spectral moments analysis of the LTAS as an objective test to distinguish ADSD from MTD. STUDY DESIGN: Case-control comparison. METHODS: Pretreatment voice samples from 59 subjects with MTD (10 males and 49 females) and 41 subjects with ADSD (19 males and 22 females) were analyzed. Groups were separated by gender, adjusted for age, and results from the analysis were compared across different analyzing bandwidths. Diagnostic precision estimates were calculated including sensitivity, specificity, positive and negative predictive values, and positive and negative likelihood ratios. RESULTS: No significant spectral differences were found between men in both groups. However, significant between-group differences were identified for all spectral moments for women. Logistic stepwise regression identified that spectral standard deviation (moment 2) uniquely distinguished women with MTD and ADSD. No other spectral moments contributed significant discriminatory information. CONCLUSIONS: The results suggest that moment 2 of the LTAS provides respectable diagnostic precision by highlighting spectral noise differences between females with MTD and ADSD. Automated spectral moments analysis deserves further attention as a possible test for differential diagnosis.