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.

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.

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-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.

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 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.

Multi-Institutional Study of Patient-Reported Outcomes of Paradoxical Vocal Fold Motion.

OBJECTIVE: To explore patient-reported outcome measures of pediatric paradoxical vocal fold motion through a multi-institutional study of geographically diverse United States medical facilities to assess long-term management and outcomes. METHODS: Eligible participants >8 years of age diagnosed with PVFM over a 10-year period from 7 tertiary pediatric hospitals were invited to complete a survey addressing study objectives. RESULTS: 65 participants completed the survey, of whom 80% were female, 75% reported a 3.5 grade point average or better, and 75% identified as competitive athletes or extremely athletic individuals. Participants rated their perceived efficacy of 13 specific treatments. Only five treatments were considered effective by a majority of the participants who tried them. The treatments that participants tried most often were breathing exercises (89.2%), bronchodilator treatments (45%), and allergy medications (35.4%). 78.8% of participants reported receiving more than one treatment and 25% reported receiving a combination of bronchodilators, anticholinergics, and steroids. At the time of PVFM diagnosis, 38% of participants had no idea when their symptoms would completely resolve. 23.3% of participants did not experience symptom resolution until greater than 1 year after diagnosis. CONCLUSIONS: Traditional management tools such as breathing exercises and biofeedback treatments may not provide the long-term benefit that providers anticipate. In addition to these commonly used management strategies, highly efficacious techniques such as counseling and lifestyle management should be incorporated into the long-term management of patients whose symptoms are refractory to traditional care. LEVEL OF EVIDENCE: 4 Laryngoscope, 133:970-976, 2023.

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.

Computed Tomography Imaging of the Larynx for Diagnosis of Vocal Cord Dysfunction.

BACKGROUND: Vocal cord dysfunction/inducible laryngeal obstruction (VCD/ILO) is characterized by breathlessness and often mimics or accompanies severe asthma. The disorder occurs intermittently, and the diagnosis is established by using laryngoscopy. Dynamic computed tomography (CT) imaging of the larynx at low-radiation doses has the potential to provide an alternative method to make the diagnosis of VCD/ILO. METHODS: We report two case series: in series A, laryngoscopy (diagnostic standard) and CT imaging of the larynx were each performed within 1 hour of each other (n=31), and in series B, the procedures were performed on separate days 4 to 6 weeks apart (n=72). Diagnosis of VCD/ILO by laryngoscopy used conventional criteria, and diagnosis by CT imaging was based on vocal cord narrowing in excess of a validated normal threshold. In each series, we evaluated the accuracy of CT imaging of the larynx to establish a diagnosis of VCD/ILO compared with laryngoscopy. RESULTS: In series A, the sensitivity of CT imaging of the larynx was 53.8%, and specificity was 88.9%; in series B, the sensitivity of CT imaging of the larynx was 76.2%, and specificity was 93.3%. At a disease prevalence of 30% (which was known to be the case in our clinic), the positive predictive value was 67.5% in series A and 83% in series B. Negative predictive values were 81.8% and 90.1% in series A and B, respectively, and false-positive rates were 11.1% and 6.7%. CONCLUSIONS: When the population prevalence was assumed to be 30%, low-dose CT imaging of the larynx detected VCD/ILO with negative predictive values greater than 80% in both series settings and agreed with each other within 9 percentage points. Positive predictive values for laryngeal CT imaging varied substantially between the settings of the two case series. (Supported by Monash Lung and Sleep Institute and Grant APP ID 1198362 and others.)

Diagnosis of vocal cord dysfunction / inducible laryngeal obstruction-A Delphi study protocol.

INTRODUCTION: Currently there is no consistent and widely accepted approach to the diagnosis of vocal cord dysfunction/inducible laryngeal obstruction (VCD/ILO). Harmonised diagnostic methods are vital to enable optimal diagnosis, advance management and enable research. We aim to obtain consensus on how expert clinicians recognise and diagnose VCD/ILO. METHODS AND ANALYSIS: Two-round modified Delphi, with workshop validation. ETHICS AND DISSEMINATION: Institutional Board Review was obtained from the Monash Health Human Research Ethics Committee. The dissemination plan is for presentation and publication. REGISTRATION DETAILS: Registered at Australia and New Zealand Clinical Trials Registry ACTRN12621001520820p.

[Differential diagnosis of asthma and dysfunctional breathing and vocal cord dysfunction].

The clinical manifestations of bronchial asthma are complex and variable, which can easily be confused with other diseases. So misdiagnosis and missed diagnosis of asthma are common. In the differential diagnosis of asthma, organic diseases such as left ventricular dysfunction, chronic obstructive pulmonary disease, and upper airway obstruction are most often considered, but the understanding of functional disease is insufficient. In fact, GINA guidelines suggested that the differential diagnosis in adult patients with suspected asthma needed to prioritize functional diseases such as dysfunctional breathing (DB)/hyperventilation syndrome (HVS) and vocal cord dysfunction (VCD), rather than organic disease. Here we review the concepts of DB and VCD and their differentiation from asthma.

Clinical presentation of paradoxical vocal fold motion or laryngeal dyskinesia in infants.

OBJECTIVES: Paradoxical vocal fold motion (PVFM) is not well-characterized in infants. Sex- and race/ethnicity-based differences have been described in older children with PVFM. This study's objectives are to characterize demographic and clinical characteristics of infants diagnosed with PVFM and investigate sex- and race-specific differences in presentation. METHODS: We retrospectively reviewed infants ≤1 year of age diagnosed with PVFM at our institution from 2009 to 2019. Patient demographics, symptoms, and findings on flexible laryngoscopy are described. Sex- and race/ethnicity-based differences were assessed using Fisher's exact test analyses. RESULTS: We identified 22 infants who were diagnosed with PVFM. The average age (range) at diagnosis was 5.7 (0.25-12.0) months, and 45.5% were male. The majority (54.6%) of patients identified as non-Hispanic White. Common comorbidities included GERD (45.5%) and chronic rhinitis (13.6%). Stridor was the only presenting symptom in the majority of patients (95.4%). The most common episode triggers were crying (45.5%), feeding (27.3%), and gastric reflux (9.1%). On flexible laryngoscopy, PVFM was observed in 95.5% of patients. A third of patients (31.8%) were misdiagnosed as having reactive airway disease or laryngomalacia prior to evaluation by otolaryngology. No sex- and race/ethnicity-based differences in presentation were identified. CONCLUSION: We present the largest case series of PVFM in infants. We found sparse clinical signs/symptoms other than stridor and a high incidence of misdiagnosis, which supports the importance of objective flexible laryngoscopy for the evaluation of stridor in this age group. Previously reported sex- and race/ethnicity-based differences in presentation of PVFM were not observed in this cohort of infants.

The impact of a fast-track questionnaire in pediatric paradoxical vocal fold motion disorder (PVFMD).

OBJECTIVE: PVFMD is a frequent cause of dyspnea in the healthy adolescent. When PVFMD is suspected, the current standard of care includes referral to an otolaryngologist (ENT) prior to beginning laryngeal control therapy (LCT) with a speech language pathologist (SLP). We hypothesize that a "fast-track" screening questionnaire will improve time to treatment and decrease patient charges. METHODS: Patients (n = 258, group 1) who received traditional referral and were evaluated in pediatric voice clinic with a diagnosis of PVFMD between 11/2013 and 11/2017 were identified and compared with 66 patients (group 2) from 10/2018 to 11/2019 who were prospectively studied and placed into a fast-tracked subgroup for LCT without preceding ENT evaluation if they scored 8/10 or higher on a designed screening questionnaire. RESULTS: Female gender (group 1: 81%, group 2: 83%, p = 0.73) and median age (group 1:14 years IQR 4; group 2:14 years IQR 3, p = 0.83) were similar. The median duration from symptom onset to LCT was shorter for group 2 (group 1: 12 months, IQR 18; group 2: 8.5 months, IQR 8)(p = 0.02). Time from referral to LCT was shorter for group 2 at 3 weeks (IQR 3) compared to group 1 at 4 weeks (IQR 3.5, p < 0.01). The minimum single patient charge for group 1 was estimated at $5123 and $1649 for group 2, yielding a potential reduction of charges of over $3000. CONCLUSION: Using a fast-track screening questionnaire for pediatric PVFMD patients significantly decreases the time to treatment without altering the response rate of LCT.

Factors impacting therapy duration in children and adolescents with Paradoxical Vocal Fold Movement (PVFM).

PURPOSE: Paradoxical Vocal Fold Movement (PVFM) may cause airway restriction and resulting dyspnea in the pediatric population. Therapy with a speech-language pathologist (SLP) is the primary treatment for children and adolescents diagnosed with Paradoxical Vocal Fold Movement (PVFM). This study examined treatment duration and factors predicting number of therapy sessions required. METHODS: Data were drawn from the University of Wisconsin-Madison Voice and Swallow Clinics Outcome Database. One hundred and twelve children and adolescents were included in this study. Participants were diagnosed with PVFM, followed for therapy with a SLP, and were subsequently discharged from therapy with successful outcomes. Extracted data included number of therapy sessions, PVFM symptoms, patient demographics, medical history, and comorbid diagnoses. Regression was used to determine factors predicting therapy duration. RESULTS: Patients completed an average of 3.4 therapy sessions before discharge. Comorbid behavioral health diagnosis (ß = 1.96, t = 3.83, p < .01) and a history of upper airway surgeries (ß = 1.26, t = 2.615, p = .01) were significant predictors of the number of therapy sessions required before discharge; both factors significantly increased therapy duration. Age, symptom trigger-type, reflux symptoms, and dysphonia did not predict therapy duration. Overall, our regression model accounted for 42% of the variance in number of sessions required (r2 = 0.42). CONCLUSIONS: On average, 3.4 sessions of therapy with an SLP resolved PVFM symptoms. Children with a behavioral health diagnosis required an average of 5.45 sessions and those with a history of upper airway surgery an average of 4.3 sessions. Future work should examine the relationship between behavioral health care and PVFM treatment, as well as how PVFM treatment efficiency can be maximized.