Bilateral Vocal Fold Abduction Dysfunction: A Report of Two Neonatal Cases.

Stridor is caused by oscillation of the narrowed upper airway. The most common cause of neonatal stridor is laryngomalacia, followed by vocal fold abduction dysfunction. Herein, we present two neonatal cases of idiopathic dysfunction of vocal fold abduction. A neonate was admitted to the neonatal intensive care unit (NICU) on day 4 of life for inspiratory stridor, intermittent subcostal retraction, and cyanosis. A second neonate was admitted to the NICU on day 7 of life for inspiratory stridor and cyanosis when crying. Neither patient had dysmorphic features or unusual cardiac ultrasonography findings. The diagnosis was confirmed by laryngo-bronchoscopy. Conservative treatment with biphasic positive airway pressure was effective in both cases and symptoms resolved within a few months. Resolution of vocal fold abduction dysfunction was confirmed by repeat endoscopy. Clinical manifestations of vocal fold abduction dysfunction vary widely. Although most cases resolve spontaneously, prolonged tube feeding, or even tracheostomy, is needed in some severe cases. Diagnosis of vocal fold abduction dysfunction requires a laryngo-bronchoscopy study; thus, there may be a large number of undiagnosed patients. Vocal fold abduction dysfunction should be considered in the differential diagnosis for neonatal inspiratory stridor.

Exploring Mental Health in a Pediatric Paradoxical Vocal Fold Motion Sample Using Patient-Reported Outcomes.

OBJECTIVE: Paradoxical vocal fold motion (PVFM) is characterized by inappropriate adduction of vocal folds during inspiration causing dyspnea. While anxiety is suspected to be a predisposing factor, incidence has been understudied. STUDY DESIGNS: Retrospective review. SETTING: Multidisciplinary PVFM hospital clinic. METHODS: We used patient-reported outcome measures to examine anxiety and depression in consecutive patients aged 10 to 17 years using Pediatric SFv1.1 Anxiety 8b and Level 2-Depression inventories (parents completed proxy forms). T-scores were classified as normal (none to slight <55) or elevated (mild 55-59.9, moderate 60-69.9, severe >70). RESULTS: Twenty-three pediatric patients and 20 parents completed surveys. Mean age was 13.74 years. For anxiety, 69.6% of patients and 40% of parents identified elevated levels. For depression, 30.4% of patients and 15% of parents identified elevated levels. Therapy need for the sample was 65.2% (34.8% active in services and 30.4% referred). Child anxiety scores were significantly higher in the therapy need group, U = 17, P = .004. CONCLUSION: This study of adolescents with PVFM confirmed elevated anxiety and depression scores in 2/3 of the participants. Anxiety likely precedes diagnosis and is a predisposing factor. Referral for individualized intervention targeting anxiety and depression is indicated.

Living With Asthma and Vocal Cord Dysfunction/Inducible Laryngeal Obstruction: "I Just Can't Get Air In".

BACKGROUND: Asthma and vocal cord dysfunction (VCD), also known as inducible laryngeal obstruction (ILO), may coexist, resulting in worse outcomes for patients. The experience of people with VCD/ILO and coexisting asthma is unknown. OBJECTIVE: We sought to determine whether coexistent VCD/ILO and asthma have deleterious impacts on quality of life. METHODS: We undertook a descriptive qualitative study using one-to-one semistructured interviews with 30 purposively recruited adult participants with a prior confirmed doctor asthma diagnosis and laryngoscopy-confirmed VCD/ILO. A thematic and content analysis was conducted to explore the data. RESULTS: Participants were mostly female (63%), mean ± SD age 63 ± 12 years. Four themes were identified: trapped voice, altered life, knowledge about VCD/ILO, and looking for solutions. Participants reported their voice being trapped in their throat or the voice being suddenly cut off when talking or singing. Self-reported VCD/ILO symptoms including throat tightness and breathlessness were highlighted by participants. The second theme described how patients struggle to communicate or tended to shorten conversations. Insufficient knowledge and existing confusion regarding whether asthma was causing the breathlessness was described in the third theme. Looking for solutions depicted participants' diagnostic journey and how they sought an explanation for the symptoms. CONCLUSIONS: People with asthma and coexisting VCD/ILO experience a substantial burden affecting the quality of life. These data describe the impact on patients with coexisting conditions and should be used to increase clinician awareness of the experience of VCD/ILO from patients' perspectives to support a personalized approach to care.

Protocol for a scoping review to map health outcomes in individuals with inducible laryngeal obstruction.

BACKGROUND: Inducible laryngeal obstruction causes narrowing of the laryngeal aperture in response to external triggers. Outcomes are measured in inducible laryngeal obstruction to monitor changes in health status over time. METHODS: This study is a scoping review based on Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) methodology. The review will be guided by the following research question: 'What health outcomes are measured in studies including people with inducible laryngeal obstruction?' The research question was validated using the Population-Concept-Context framework according to the methodology for Joanna Briggs Institution Scoping Reviews. Relevant peer-reviewed studies and grey literature conducted over the last 40 years will be identified from electronic databases including AMED, CINAHL, Embase, EMCARE, MEDLINE, OVID, PubMed and PsycINFO. The search strings 'inducible laryngeal obstruction', 'ILO', 'vocal cord dysfunction', 'VCD', 'paradoxical vocal fold motion', 'PVFM', 'outcome', 'measure', 'measurement instrument', 'assessment', 'scale', 'questionnaire' will be combined using Boolean logic. An independent reviewer will conduct title screening; two independent reviewers will conduct abstract and full article screening, followed by data extraction by two reviewers. Analyses will be conducted appropriate to the findings. DISCUSSION: The review will document evidence of health outcomes measured in inducible laryngeal obstruction, identifying measurement characteristics and potential utility. Collating studies may identify gaps in coverage, the need for novel tools, and for standardisation for clinical and research purposes. WHAT THIS PAPER ADDS: What is already known on the subject Inducible laryngeal obstruction causes narrowing of the laryngeal aperture in response to external triggers. Outcomes are measured in inducible laryngeal obstruction to monitor changes in health status over time. Currently, there are no standardised outcome measures for measuring the effects of interventions in inducible laryngeal obstruction (ILO). What this paper adds to existing knowledge Assessment of health can be measured in a variety of ways. Physiological, radiological and biochemical measurements of impairment are more common historically but there are a lot of outcomes of other factors now including subjective measures of functional status and health-related quality of life, with data collected directly from patients. This study will allow us to scope the literature to see the health outcomes being measured in ILO to attempt to standardise and develop future health outcomes. What are the potential or actual clinical implications of this work? The review will document evidence of health outcomes measured in inducible laryngeal obstruction, identifying measurement characteristics and potential utility. Collating studies may identify gaps in coverage, the need for novel tools and for standardisation for clinical and research purposes.

Vocal cord dysfunction causing hypoxaemia in the postanaesthesia care unit.

Hypoxaemia in the postanaesthesia care unit is common and the majority is caused by hypoventilation or upper airway obstruction due to the (residual) effects of anaesthetic and analgesic agents. We present a case of upper airway obstruction caused by vocal cord dysfunction, a less frequently occurring aetiology. The patient's case suggests a notable relationship between procedural laryngeal stimulus and the onset of symptoms. Approach to the diagnosis and flexible laryngoscopy to either rule-in or rule-out several relevant differentials are discussed.

Nerve monitoring in endocrine surgery: Practice patterns differ among surgeons for parathyroidectomy and thyroidectomy.

BACKGROUND: It is unknown whether intraoperative nerve monitoring is associated with reduced vocal cord dysfunction after parathyroidectomy. We aimed to investigate intraoperative nerve monitoring use among Collaborative Endocrine Surgery Quality Improvement Program surgeons and factors associated with vocal cord dysfunction after parathyroidectomy. METHODS: Patients who underwent parathyroidectomy included in the Collaborative Endocrine Surgery Quality Improvement Program (2014-2022) were identified. The annual percent change in parathyroidectomies performed with intraoperative nerve monitoring was calculated using joinpoint regression. Multivariable logistic regression was used to compare outcomes between patients undergoing parathyroidectomy with/without intraoperative nerve monitoring. To compare surgeon-specific trends, Collaborative Endocrine Surgery Quality Improvement Program thyroidectomy and parathyroidectomy datasets (2014-2021) were combined. Parathyroidectomies performed by surgeons who used intraoperative nerve monitoring consistently in thyroidectomy were identified. Factors associated with intraoperative nerve monitoring were examined using multivariable logistic regression. RESULTS: A total of 9,813 patients underwent parathyroidectomy. Intraoperative nerve monitoring was used in 49% of cases (n = 4,818). There was an increase in parathyroidectomies with intraoperative nerve monitoring from 2014 to 2018 (annual percent change 22.2, P = .01), followed by a plateau (2018-2022 annual percent change -0.66, P = .85). Few patients (0.44%, n = 43) developed vocal cord dysfunction. Vocal cord dysfunction was not associated with intraoperative nerve monitoring (adjusted odds ratio 0.92, P = .75). Whereas 41% (n = 56/138) of surgeons used intraoperative nerve monitoring routinely in parathyroidectomy, 65% (n = 90/138) used it routinely in thyroidectomy. Among surgeons who used intraoperative nerve monitoring routinely in thyroidectomy, only 57% used it routinely in parathyroidectomy; factors associated with intraoperative nerve monitoring during parathyroidectomy included reoperation (adjusted odds ratio 2.51, P < .01), secondary/tertiary hyperparathyroidism (adjusted odds ratio 1.42, P = .02), multiglandular disease (adjusted odds ratio 1.76, P < .001), and non-localized disease (adjusted odds ratio 1.65, P < .001). CONCLUSION: Endocrine surgeons use intraoperative nerve monitoring selectively. Surgeons who routinely use intraoperative nerve monitoring during thyroidectomy are more likely to use it during parathyroidectomy. Future studies should determine who may benefit most from intraoperative nerve monitoring in parathyroidectomy.

Routine use of intraoperative nerve monitoring is associated with a reduced risk of vocal cord dysfunction after thyroid cancer surgery.

BACKGROUND: The aim of this study was to investigate the associations between individual surgeon's intraoperative nerve monitoring (IONM) practice and factors associated with vocal cord (VC) dysfunction in patients with thyroid cancer undergoing thyroidectomy. METHODS: Using Collaborative Endocrine Surgery Quality Improvement Program (CESQIP) 2014-21 data, multivariable logistic regression analyses investigated variables associated with short- and long-term VC-dysfunction, associations of routine use of IONM with postoperative outcomes, and patient characteristics associated with IONM use. RESULTS: Among 5,446 patients (76.7% female, mean age 49 years), 68.5% had surgery by surgeons using IONM in ≥ 90% of cases (63% of surgeons, n = 73). Post-operative VC-dysfunction was diagnosed by laryngoscopy in 3.0% of patients in the short-term and 2.7% in the long-term. When surgeons routinely used IONM, the incidence of VC-dysfunction was 2.4% in the short-term and 2.2% in the long-term, compared to 4.4% and 3.7%, respectively, when surgeons did not routinely use IONM (p < 0.01). After adjustment, routine use of IONM was independently associated with reduced risk of short- (OR 0.48, p < 0.01) and long-term (OR 0.52, p < 0.01) VC-dysfunction, a lower risk of postoperative hypoparathyroidism in the short- (OR 0.67, p < 0.01) and long-term (OR 0.54, p < 0.01), and higher likelihood of same-day discharge (OR 2.03, p < 0.01). Extrathyroidal tumor extension and N1-stage were factors associated with postoperative VC-dysfunction in the short- (OR 3.12, p < 0.01; OR 1.92, p = 0.01, respectively) and long-term (OR 3.11, p < 0.01; OR 2.32, p < 0.01, respectively). CONCLUSION: Routine use of IONM was independently associated with a lower risk of endocrine surgery-specific complications and greater likelihood of same-day discharge.

Diagnosis of vocal cord dysfunction/inducible laryngeal obstruction: An International Delphi Consensus Study.

BACKGROUND: Vocal cord dysfunction/inducible laryngeal obstruction (VCD/ILO) is characterized by breathing difficulties in association with excessive supraglottic or glottic laryngeal narrowing. The condition is common and can occur independently; however, it may also be comorbid with other disorders or mimic them. Presentations span multiple specialties and misdiagnosis or delayed diagnosis is commonplace. Group-consensus methods can efficiently generate internationally accepted diagnostic criteria and descriptions to increase clinical recognition, enhance clinical service availability, and catalyze research. OBJECTIVES: We sought to establish consensus-based diagnostic criteria and methods for VCD/ILO. METHODS: We performed a modified 2-round Delphi study between December 7, 2021, and March 14, 2022. The study was registered at ANZCTR (Australian New Zealand Clinical Trials Registry; ACTRN12621001520820p). In round 1, experts provided open-ended statements that were categorized, deduplicated, and amended for clarity. These were presented to experts for agreement ranking in round 2, with consensus defined as ≥70% agreement. RESULTS: Both rounds were completed by 47 international experts. In round 1, 1102 qualitative responses were received. Of the 200 statements presented to experts across 2 rounds, 130 (65%) reached consensus. Results were discussed at 2 international subject-specific conferences in June 2022. Experts agreed on a diagnostic definition for VCD/ILO and endorsed the concept of VCD/ILO phenotypes and clinical descriptions. The panel agreed that laryngoscopy with provocation is the gold standard for diagnosis and that ≥50% laryngeal closure on inspiration or Maat grade ≥2 define abnormal laryngeal closure indicative of VCD/ILO. CONCLUSIONS: This Delphi study reached consensus on multiple aspects of VCD/ILO diagnosis and can inform clinical practice and facilitate research.

Vocal cord dysfunction/inducible laryngeal obstruction: novel diagnostics and therapeutics.

INTRODUCTION: Vocal cord dysfunction/inducible laryngeal obstruction (VCD/ILO) is an important medical condition but understanding of the condition is imperfect. It occurs in healthy people but often co-exists with asthma. Models of VCD/ILO pathophysiology highlight predisposing factors rather than specific mechanisms and disease expression varies between people, which is seldom appreciated. Diagnosis is often delayed, and the treatment is not evidence based. AREAS COVERED: A unified pathophysiological model and disease phenotypes have been proposed. Diagnosis is conventionally made by laryngoscopy during inspiration with vocal cord narrowing >50% Recently, dynamic CT larynx was shown to have high specificity (>80%) with potential as a noninvasive, swift, and quantifiable diagnostic modality. Treatment entails laryngeal retraining with speech pathology intervention and experimental therapies such as botulinum toxin injection. Multidisciplinary team (MDT) clinics are a novel innovation with demonstrated benefits including accurate diagnosis, selection of appropriate treatment, and reductions in oral corticosteroid exposure. EXPERT OPINION: Delayed diagnosis of VCD/ILO is pervasive, often leading to detrimental treatments. Phenotypes require validation and CT larynx can reduce the necessity for laryngoscopy, thereby fast-tracking diagnosis. MDT clinics can optimize management. Randomized controlled trials are essential to validate speech pathology intervention and other treatment modalities and to establish international standards of care.

Vocal cord dysfunction/inducible laryngeal obstruction-2022 Melbourne Roundtable Report.

Vocal cord dysfunction/inducible laryngeal obstruction (VCD/ILO), is a common condition characterized by breathlessness associated with inappropriate laryngeal narrowing. Important questions remain unresolved, and to improve collaboration and harmonization in the field, we convened an international Roundtable conference on VCD/ILO in Melbourne, Australia. The aims were to delineate a consistent approach to VCD/ILO diagnosis, appraise disease pathogenesis, outline current management and model(s) of care and identify key research questions. This report summarizes discussions, frames key questions and details recommendations. Participants discussed clinical, research and conceptual advances in the context of recent evidence. The condition presents in a heterogenous manner, and diagnosis is often delayed. Definitive diagnosis of VCD/ILO conventionally utilizes laryngoscopy demonstrating inspiratory vocal fold narrowing >50%. Computed tomography of the larynx is a new technology with potential for swift diagnosis that requires validation in clinical pathways. Disease pathogenesis and multimorbidity interactions are complex reflecting a multi-factorial, complex condition, with no single overarching disease mechanism. Currently there is no evidence-based standard of care since randomized trials for treatment are non-existent. Recent multidisciplinary models of care need to be clearly articulated and prospectively investigated. Patient impact and healthcare utilization can be formidable but have largely escaped inquiry and patient perspectives have not been explored. Roundtable participants expressed optimism as collective understanding of this complex condition evolves. The Melbourne VCD/ILO Roundtable 2022 identified clear priorities and future directions for this impactful condition.

Predictors of voice therapy efficacy in vocal cord dysfunction at a tertiary care center.

OBJECTIVE: Vocal cord dysfunction is inappropriate adduction of vocal cords during inspiration that causes dyspnea and is commonly mistaken for exercise-induced asthma. To improve diagnostic accuracy, this study aims to identify demographics associated with vocal cord dysfunction and to determine their impact on the efficacy of voice therapy in improving vocal cord function. STUDY DESIGN: Retrospective chart review. SETTING: Single tertiary care institution between January 2015 and December 2021. METHODS: 184 patients who underwent voice therapy for vocal cord dysfunction were included. The primary outcome was patient self-reported percent improvement of symptoms. The secondary outcome was number of voice therapy treatments. RESULTS: The mean duration of symptoms was 2 ± 3 years. The mean number of voice therapy treatments was 2.2 ± 1.5. Of the 107 (58.2 %) patients with documented perceived breathing improvement percentages recorded, the mean maximal percent improvement was 72.5 ± 21.5 %. Mean maximal percent improvement of symptoms increased with each voice therapy treatment (p = 0.01). This association remained significant when controlling for comorbid conditions such as allergic rhinitis with postnasal drip, anxiety, asthma, and gastroesophageal reflux disease in multivariate analysis (p = 0.005). Patients with asthma had significantly higher maximum percent breathing improvement compared to those without asthma (p = 0.026). Similarly, patients who played sports had significantly higher maximum percent breathing improvement compared to those who did not (p = 0.022). CONCLUSION: Patient perceived breathing improvement with voice therapy is higher among those with concomitant asthma and those who play sports. Voice therapy is a safe and effective first line treatment of vocal cord dysfunction even when controlling for comorbid conditions.

Vocal Cord Dysfunction During the COVID-19 Pandemic.

BACKGROUND: Vocal cord dysfunction is an upper-airway disorder characterized by exaggerated and transient glottic constriction causing respiratory and laryngeal symptoms. Common presentation is with inspiratory stridor often in the context of emotional stress and anxiety. Other symptoms include wheezing (which may be on inspiration), frequent cough, choking sensation, or throat and chest tightness. This is seen commonly in teenagers, particularly in adolescent females. The COVID-19 pandemic has been a trigger for anxiety and stress with an increase in psychosomatic illness. Our objective was to find out if the incidence of vocal cord dysfunction increased during COVID-19 pandemic. METHODS: We performed a retrospective chart review of all the subjects with a new diagnosis of vocal cord dysfunction who were seen at the out-patient pulmonary practice at our children's hospital between January 2019-December 2020. RESULTS: The incidence of vocal cord dysfunction in 2019 was found to be 5.2%, (41/786 subjects seen) compared to 10.3% (47/457 subjects seen) in 2020, which is a nearly 100% increase in incidence (P < .001). CONCLUSIONS: It is important to recognize that vocal cord dysfunction has increased during the COVID-19 pandemic. In particular, physicians treating pediatric patients, as well as respiratory therapists, should be aware of this diagnosis. It is imperative to avoid unnecessary intubations and treatments with bronchodilators and corticosteroids as opposed to behavioral and speech training to learn effective voluntary control over the muscles of inspiration and the vocal cords.

Inducible Laryngeal Obstruction Differential Diagnosis in Adolescents and Adults: A Tutorial.

Inducible laryngeal obstruction (ILO), formerly referred to as paradoxical vocal fold motion and vocal cord dysfunction, is a complex disorder of the upper airway that requires skillful differential diagnosis. There are several medical conditions that may mimic ILO (or which ILO may mimic) that should be considered in the differential diagnosis before evidence-supported behavioral intervention is initiated to mitigate or eliminate this upper airway condition. A key in treatment planning is determination of an isolated presentation of ILO or ILO concurrent with other conditions that affect the upper airway. Accurate, timely differential diagnosis in the clinical assessment of this condition mitigates delay of targeted symptom relief and/or insufficient intervention. Accurate assessment and nuanced clinical counseling are necessary to untangle concurrent, competing conditions in a single patient. This tutorial describes the common and rare mimics that may be encountered by medical professionals who evaluate and treat ILO, with particular attention to the role of the speech-language pathologist. Speech-language pathologists receive referrals for ILO from several different medical specialists (allergy, pulmonology, and sports medicine), sometimes without a comprehensive team assessment. It is paramount that speech-language pathologists who assess and treat this disorder have a solid understanding of the conditions that may mimic ILO. Summary tables that delineate differential diagnosis considerations for airway noise, origin of noise, symptoms, triggers, role of the speech-language pathologist, and ß-agonist response are included for clinician reference. A clinical checklist is also provided to aid clinicians in the critical assessment process.

Tamoxifen Alters TGF-ß1/Smad Signaling in Vocal Fold Injury.

OBJECTIVES: Effective treatments for vocal fold fibrosis remain elusive. Tamoxifen (TAM) is a selective estrogen receptor modulator and was recently reported to have antifibrotic actions. We hypothesized that TAM inhibits vocal fold fibrosis via altered transforming growth factor beta 1 (TGF-ß1) signaling. Both in vitro and in vivo approaches were employed to address this hypothesis. METHODS: In vitro, vocal fold fibroblasts were treated with TAM (10-8 or 10-9 M) ± TGF-ß1 (10 ng/ml) to quantify cell proliferation. The effects of TAM on genes related to fibrosis were quantified via quantitative real-time polymerase chain reaction. In vivo, rat vocal folds were unilaterally injured, and TAM was administered by oral gavage from pre-injury day 5 to post-injury day 7. The rats were randomized into two groups: 0 mg/kg/day (sham) and 50 mg/kg/day (TAM). Histological changes were examined on day 56 to assess tissue architecture. RESULTS: TAM (10-8 M) did not affect Smad3, Smad7, Acta2, or genes related to extracellular matrix metabolism. TAM (10-8 or 10-9 M) + TGF-ß1, however, significantly increased Smad7 and Has3 expression and decreased Col1a1 and Acta2 expression compared to TGF-ß1 alone. In vivo, TAM significantly increased lamina propria area, hyaluronic acid concentration, and reduced collagen deposition compared to sham treatment. CONCLUSIONS: TAM has antifibrotic potential via the regulation of TGF-ß1/Smad signaling in vocal fold injury. These findings provide foundational data to develop innovative therapeutic options for vocal fold fibrosis. LEVEL OF EVIDENCE: NA Laryngoscope, 133:2248-2254, 2023.

Characterizing Pediatric Bilateral Vocal Fold Dysfunction: Analysis with the Pediatric Health Information System Database.

OBJECTIVES: The purpose of this study was to characterize pediatric bilateral vocal fold dysfunction and to examine the overall inpatient mortality. METHODS: Retrospective cohort analysis. Data from the Pediatric Health Information System was gathered for all pediatric patients with a diagnosis of bilateral vocal fold dysfunction between January 2008 and September 2020. Univariate and multivariate analyses were performed using Cox proportional hazard models. RESULTS: 2395 patients accounted for 4799 hospitalizations with bilateral vocal fold dysfunction. Inpatient mortality occurred in 2.9% of the study sample. Chiari 2 was found in 2.8% of patients. The most common associated diagnoses were related to comorbid respiratory conditions (61.1%). The median adjusted ratio of cost to charges was $76,569. Aspiration was noted in 28 patients (1.2%). Gastrostomy was performed in 607 patients (25.3%). Tracheostomy was performed in 27% of patients. The overall 90-day readmission rate was 61%. On multivariate analysis, prognostic factors associated with increased hospital survival include gastrointestinal comorbidities (hazard ratio [HR]: 0.29; 95% confidence interval [CI]: 0.18-0.49) and tracheostomy (HR: 0.21; 95% CI: 0.12-0.37). CONCLUSION: This database study represents the largest cohort analysis to date characterizing bilateral vocal fold dysfunction. Favorable prognostic indicators of overall hospital survival include gastrointestinal comorbidities and the presence of tracheostomy. Tracheostomy is associated with an increase in hospital costs, comorbidities, gastrostomy tube placement, and Chiari diagnosis. LEVEL OF EVIDENCE: 4 Laryngoscope, 133:1228-1233, 2023.