Functional Electrical Stimulation of the Feline Larynx With a Flexible Ribbon Electrode Array.

OBJECTIVES: Success of laryngeal reanimation through neurorrhaphy has been limited by synkinesis and preoperative muscle atrophy. The objective of this study was to investigate the use of epimysial electrode arrays as a means of delivering electrical stimulation to the posterior cricoarytenoid muscles in order to control laryngeal abduction. METHODS: Ribbon electrode arrays with 4 or 8 electrode contacts were used. Four cats underwent implantation of electrode arrays along the surface of the posterior cricoarytenoid muscles. The glottis was visualized with a 0° telescope while electrodes were stimulated at different amplitudes and pulse-width durations. Recordings of stimulated vocal folds were analyzed, and the degree of vocal fold abduction was measured in order to create recruitment curves for the left and right posterior cricoarytenoid. Recruitment curves from electrode channels within the array were compared. RESULTS: Electrodes oriented along the medial aspect of the posterior cricoarytenoid stimulated graded physiologic degrees of abduction depending on the amplitude of stimulation. Electrodes oriented laterally along the posterior cricoarytenoid stimulated greater degrees of simultaneous adduction with abduction. CONCLUSION: Acute studies of ribbon surface electrode arrays placed onto the posterior cricoarytenoid reproduce graded degrees of abduction necessary for the precise function of respiration and speech.

Type II Thyroplasty with Bilateral Partial Cricothyroid Myectomy for Vocal Tic in Child with Cerebral Dysgenesis.

Vocal tics can occur in neuropsychiatric disorders and result in familial distress. Management is challenging, particularly in children with developmental delay. A 5-year-old with cerebral dysgenesis presented with a high amplitude, high-frequency vocal tic. Type II thyroplasty with bilateral cricothyroid muscle myectomy was performed after initial botulinum toxin trial. Amount, volume, and pitch of tics significantly decreased, without change in swallow. Benefits persisted at 1-year follow-up. This is the first description of combined type II thyroplasty with cricothyroid myectomy for high-frequency vocal tic. This can be done safely and provide sustained benefit for a rare, impactful voice disorder.

Long-Term Outcomes and Revision Rates in Laryngeal Reinnervation.

OBJECTIVES: Nonselective laryngeal reinnervation is an effective procedure to improve voice quality after unilateral vocal fold paralysis. Few studies have captured long-term outcome data, and the revision rate for this operation is currently unknown. The objective of this study is to describe the long-term outcomes and revision rates of unilateral, nonselective reinnervation in pediatric and adult patients. METHODS: Patients who underwent laryngeal reinnervation from 2000 to 2022 with a single surgeon were identified for inclusion. Patients who underwent bilateral, super selective, deinnervation and reinnervation, and/or concurrent arytenoid adduction procedures were excluded. Outcome measures included maximum phonation time [MPT], voice handicap index score [VHI], patient-reported percent normal voice, revision procedures, and complications. Data were compiled and analyzed using paired t-tests, repeated measures analysis of covariance, and binary logistic regression analysis. RESULTS: One hundred thirty-two patients underwent unilateral, nonselective ansa-recurrent laryngeal nerve [RLN] laryngeal reinnervation. Reinnervation significantly improved MPT and patient-reported percentage of normal voice and significantly decreased VHI. Eleven patients underwent revision procedures, corresponding to a revision rate of 8.3%. Additional procedures included medialization laryngoplasty [n = 3], medialization laryngoplasty with arytenoid adduction [n = 3] and injection augmentation greater than 1 year after reinnervation [n = 5]. The only factor associated with the need for additional surgery was time lapse from nerve injury to reinnervation. The overall complication rate was 6.8%; no patient required reintubation or tracheostomy. CONCLUSION: Unilateral, nonselective laryngeal reinnervation can provide reliable improvement in vocal symptoms after recurrent laryngeal nerve injury. The revision rate after laryngeal reinnervation is favorable and comparable to framework surgery. LEVEL OF EVIDENCE: 4 Laryngoscope, 134:3187-3192, 2024.

Predominantly unilateral laryngomalacia in infants with unilateral vocal fold paralysis.

BACKGROUND: Neonatal unilateral vocal fold paralysis may arise iatrogenically, idiopathically, or in the context of an underlying neurologic disorder. Management is often supportive, focusing on diet modification to allow for safe oral feeding. We describe the clinical course of six infants with unilateral vocal fold paralysis who developed predominantly unilateral laryngomalacia ipsilateral to the affected vocal fold with associated severe respiratory symptoms and feeding difficulty. METHODS: Retrospective review of six infants with unilateral vocal fold paralysis and predominantly unilateral laryngomalacia. Charts were reviewed for etiology of vocal fold paralysis, presenting symptoms, operative details, postoperative course, and outcomes for breathing and swallowing. RESULTS: Etiology of vocal fold paralysis included cardiac surgery in four patients, intubation-related in one, and idiopathic in one. Presenting symptoms included increased work of breathing, stridor, feeding difficulty, respiratory failure requiring noninvasive respiratory support, and weak cry. All infants were on nasogastric tube feedings. Direct microlaryngoscopy with unilateral or predominantly unilateral (conservative contralateral aryepiglottic fold division) supraglottoplasty was performed. Stridor and work of breathing improved in all six patients within 1 week postoperatively. Oral feeding improved in three patients within 2 weeks. Three patients had persistent feeding impairment with improvement within one year. CONCLUSIONS: Predominantly unilateral laryngomalacia may arise in the context of unilateral vocal fold paralysis. Addressing the ipsilateral cuneiform collapse can improve breathing and feeding. This may be an under-described phenomenon and represents an additional reason to include the otolaryngologist early in the care of infants with suspected possible new unilateral vocal fold paralysis. Breathing and swallow can improve post-operatively, but feeding may remain limited by the vocal fold paralysis and any medical comorbidities. Ongoing follow-up and collaboration with speech-language pathology to optimize feeding are important.

Care of the child's voice: a pediatric otolaryngologist's perspective.

Children with voice problems are encountered by speech-language pathologists working in schools but not with sufficient frequency to yield a great experience in managing them. Their evaluation and treatment now are best handled by a team of specialists in pediatric otolaryngology and speech-language pathology with expertise in voice and resonance disorders. The voice team uses a variety of assessment tools, including perceptual, parent- and patient-based, instrumental, and imaging measures. The most important component of the diagnostic assessment is laryngoscopy in the awake child, to visualize the vocal folds while the child phonates. This review covers the pediatric otolaryngologist's approach to dysphonia in children. The common causes of dysphonia are reviewed, including vocal nodules and other lesions, reflux laryngitis, laryngeal paralysis, congenital glottic webs, laryngeal papillomatosis, functional voice disorders, and velopharyngeal incompetence. Current treatment options are continually being modified. These are discussed by highlighting recently published research.

Surgical vs interventional radiology drainage of neck abscesses in pediatric patients.

OBJECTIVE: To compare outcomes using an open surgical incision and drainage (S-ID) versus an aspiration under interventional radiology drainage (A-ID) of pediatric neck abscesses. The primary outcome measure was successful drainage requiring one intervention, and the secondary outcome measures were readmission rates and overall cost to the healthcare system. METHODS: Retrospective data collection of all pediatric patients treated at the Primary Children's Hospital for neck abscess from 2008 to 2020. Patients who did not require drainage were not included. Comparison between S-ID and A-ID was performed according to the primary and secondary outcomes. RESULTS: Two hundred fifty nine patients were included in the study. Twenty-six patients had A-IR and 234 patients had S-ID. Patient demographics were not significantly different between both groups. Overall length of stay was greater in the A-ID group (5 vs 3.3 days) (p < 0.05). A second procedure was more frequent in the A-ID group with 11/26 (42%) versus 12/234(5%) in the S-ID group (P = 0.001) even after controlling for multiple possible confounding factors. The rate of readmission was also higher in the A-ID group with 3/26 (11%) versus 7/234 (3%) in the S-ID group (P = 0.006). Both groups showed similar overall cost (P = 0.621). CONCLUSIONS: A-ID and S-ID are both methods to treat head and neck abscess requiring drainage. However, overall results indicate a higher rate of failure requiring a second intervention and a higher rate of readmission in the A-ID group. In our study cost was noted to be similar between both methods.

Long-term Outcomes of Vocal Fold Paralysis Following Patent Ductus Arteriosus Ligation in Neonates.

INTRODUCTION: In patients undergoing patent ductus arteriosus (PDA) ligation there is a significant risk of left vocal fold paralysis (LVFP) particularly in premature neonates who are small for gestational age. The objective of this study is to determine the incidence of LVFP in infants following PDA ligation and report on long-term outcomes in patients with LVFP. METHODS: We performed a prospective study of patients undergoing PDA ligation in the newborn intensive care unit (NICU) between April 2004 and May 2014. Following PDA ligation, flexible laryngoscopy was performed to assess vocal fold mobility. Patients were then followed longitudinally to determine long-term outcomes. RESULTS: A total of 163 infants underwent PDA ligation. Thirty-six patients (22%) developed LVFP following the procedure. Twenty-five percent of neonates <1500 g experienced LVFP versus 5% of patients >1500 g (p = 0.033). Patients with LVFP were more likely to require a feeding tube (64% vs. 19.6%; p < 0.05) and spent more time in the NICU (135 days vs. 106 days; p < 0.05). Twenty-four patients received long-term follow-up. Six (25%) had complete resolution of LVFP, 10 (42%) were compensated, and 8 (33%) demonstrated persistent LVFP with no improvement. CONCLUSIONS: The incidence of LVFP after PDA ligation is high especially in extremely low birth weight children. The majority of patients recovered well with time, but further surgical intervention was required in uncompensated cases. Long-term follow-up of these patients is needed to ensure improvement. Laryngoscope, 133:1257-1261, 2023.

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.

"I've never been able to burp": Preliminary description of retrograde cricopharyngeal dysfunction in children.

OBJECTIVE: Retrograde cricopharyngeal dysfunction was recently described by Bastian in 2019 and is characterized by an inability to belch, abdominal or chest pressure, odd gurgling noises, and occasional difficulty vomiting. Symptoms tend to worsen with carbonated beverages. Currently, the recommended treatment is cricopharyngeus muscle botulinum toxin injections. Prior studies have included few pediatric patients within larger datasets comprised primarily of adults. We describe our preliminary experience in pediatric patients, including presenting symptoms, treatment approach, and post-treatment outcomes. METHODS: Retrospective chart review of pediatric patients (aged <18 years) diagnosed with retrograde cricopharyngeal dysfunction based on clinical history by the senior author. Medical records were reviewed for presenting symptoms, prior testing and treatment, details of treatment, and postoperative outcomes. RESULTS: Five patients with average age of 14 ± 4 (3 females, 2 males) were included. Presenting symptoms included lifelong or nearly lifelong inability to burp (n = 5), bloating (n = 5), awkward gurgling noises (n = 3), and worsening of symptoms with carbonated beverages (n = 5). Two patients had prior normal upper endoscopy. All patients underwent cricopharyngeal botulinum toxin injection under general anesthesia, with 25-50 units of botulinum toxin injected to the posterior cricopharyngeus across 4-5 locations. All patients had resolution of symptoms with follow-up of 1.5-10 months. CONCLUSIONS: Retrograde cricopharyngeal dysfunction may be underdiagnosed due to lack of awareness of the condition. Now that the phenomenon of inability to belch has a name and is being reported in the literature, we will likely see more adult and pediatric patients with these symptoms. Pediatric patients may respond similarly to adults. Larger studies with longer-term follow-up and targeted patient-reported outcome measures are needed to characterize disease presentation and treatment outcomes.

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.

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.

Case Series of Botulinum Toxin Administered to Pregnant Patients and Review of the Literature.

Objective: To evaluate a case series of patients who received medically necessary botulinum toxin during pregnancy. Materials and Methods: Retrospective chart review of three patients who underwent repeated intralaryngeal injections of botulinum toxin during pregnancy. Chart reviews were also conducted on the children to further evaluate the safety. Results: No evidence of harm to the mothers or fetuses were found in our series, including data from pregnancy and birth records using standard measures of gestation, APGAR scores, neonatal intensive care unit stay, and time until discharge. Clinical data for 3-5 years were available for the children. No evidence of muscular weakness was noted and all diagnoses were listed. Conclusion: Botulinum toxin injection for functional airway issues was not associated with any adverse effects to the mother or fetus during pregnancy in any of the cases reviewed. We recommend further investigation to evaluate the current contraindication of elective botulinum toxin use in pregnancy.

Competency-Based Assessment Tool for Pediatric Esophagoscopy: International Modified Delphi Consensus.

OBJECTIVES/HYPOTHESIS: Create a competency-based assessment tool for pediatric esophagoscopy with foreign body removal. STUDY DESIGN: Blinded modified Delphi consensus process. SETTING: Tertiary care center. METHODS: A list of 25 potential items was sent via the Research Electronic Data Capture database to 66 expert surgeons who perform pediatric esophagoscopy. In the first round, items were rated as "keep" or "remove" and comments were incorporated. In the second round, experts rated the importance of each item on a seven-point Likert scale. Consensus was determined with a goal of 7 to 25 final items. RESULTS: The response rate was 38/64 (59.4%) in the first round and returned questionnaires were 100% complete. Experts wanted to "keep" all items and 172 comments were incorporated. Twenty-four task-specific and 7 previously-validated global rating items were distributed in the second round, and the response rate was 53/64 (82.8%) with questionnaires returned 97.5% complete. Of the task-specific items, 9 reached consensus, 7 were near consensus, and 8 did not achieve consensus. For global rating items that were previously validated, 6 reached consensus and 1 was near consensus. CONCLUSIONS: It is possible to reach consensus about the important steps involved in rigid esophagoscopy with foreign body removal using a modified Delphi consensus technique. These items can now be considered when evaluating trainees during this procedure. This tool may allow trainees to focus on important steps of the procedure and help training programs standardize how trainees are evaluated. LEVEL OF EVIDENCE: 5. Laryngoscope, 131:1168-1174, 2021.

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.

The Nerve to Thyrohyoid Muscle as a Novel Donor Nerve for Laryngeal Reinnervation.

OBJECTIVES: Recurrent laryngeal nerve (RLN) injury may be a consequence of surgical procedures of the skull base, neck, and chest, with adverse consequences to function and quality of life. Laryngeal reinnervation offers a potentially stable improvement in vocal fold position and tone. The classic donor nerve is the ansa cervicalis, but is not always available due to damage or sacrifice during previous neck surgeries. Our objective was to introduce the nerve to the thyrohyoid (TH) muscle as an alternate donor nerve for reinnervation, which has not previously been described. METHODS: Case series of two patients using the TH nerve for laryngeal reinnervation after RLN injury, with description of surgical harvest. RESULTS: Follow-up results are available for 10 months (one patient) and 3 years (one patient) demonstrating both subjective and objective improvement in function. GRBAS scores were reduced. Maximal phonation time was improved. Patient rating of voice was stable or improved postoperatively. One patient described significant preoperative dyspnea which was significantly improved postoperatively, from a score of 24 to 10 out of 40 on the dyspnea handicap index. VHI was improved in one patient, but scores elevated in the other, despite a change from "moderately severe impairment" to "normal voice" subjectively. Neither patient experienced significant complications from the procedure. CONCLUSION: Laryngeal reinnervation procedures provide good outcomes in pediatric patients. When ansa cervicalis is not available as a donor nerve, the nerve to TH provides a reasonable alternative.

Bilateral Selective Laryngeal Reinnervation for Bilateral Vocal Fold Paralysis in Children.

Importance: Bilateral vocal fold paralysis (BVFP) in pediatric patients is a challenging entity with multiple causes. Traditional approaches to managing BVFP include tracheostomy, arytenoidectomy, suture lateralization, cordotomy, and posterior cricoid enlargement. These interventions are used to create a stable airway but risk compromising voice quality. Objectives: To assess the use of bilateral selective laryngeal reinnervation (SLR) surgery to manage BVFP and restore dynamic function to the larynx in pediatric patients. Design, Setting, and Participants: In this case series performed at 2 tertiary care academic institutions, 8 pediatric patients underwent bilateral SLR to treat BVFP (5 patients with iatrogenic BVFP and 3 with congenital BVFP) from November 2004 to August 2018 with follow-up for at least 1.5 years. Interventions: Bilateral selective laryngeal reinnervation surgery. Main Outcomes and Measures: Flexible laryngoscopy findings, subjective and objective measures of voice quality, subjective swallowing function, and decannulation in patients who were previously dependent on a tracheostomy tube. Results: Participants included 6 boys and 2 girls with a median age of 9.3 (range, 2.2 to 18.0) years at the time of surgery. All 8 patients were decannulated; 6 patients had preoperative tracheostomies and 2 had perioperative tracheostomies. Voice quality, as measured using the GRBAS (grade, roughness, breathiness, asthenia, strain) scale, improved in 6 of 8 patients after reinnervation, and swallowing was not impaired in any patients. In 2 patients, GRBAS scale scores remained the same before and after surgery. Inspiratory vocal fold abduction was observed on both sides in 5 patients and on 1 side in 2 patients, with no active abduction observed in 1 patient. The follow-up period was more than 5 years in 7 of 8 patients and at least 1.5 years in all patients. Conclusions and Relevance: Bilateral SLR appears to be a promising treatment option for children with BVFP; it is currently the only option, to our knowledge, with the potential to restore abductor and adductor vocal fold movement. In patients with complete paralysis, this procedure may provide a strategy for airway management and restoration of the dynamic function of the larynx. It could be considered as a first-line technique before endolaryngeal or airway framework procedures, which carry a risk of compromising voice quality.

Morphometric Differences in the Recurrent Laryngeal Nerve in Patients with Vocal Fold Paralysis.

OBJECTIVES: Injury to the recurrent laryngeal nerve (RLN), if severe enough, can result in vocal fold paralysis. Reinnervation surgery can improve patient outcomes, but previous studies have reported a negative correlation between time since onset of paralysis and surgical outcomes. The ability of the paralyzed nerve to serve as a conduit for donor nerve fibers may be a factor in the success of reinnervation; however, changes in RLN composition after paralysis have not been well studied. Therefore, we investigated the morphometric composition of explanted RLN sections from patients who had experienced vocal fold paralysis for varying length of times. METHODS: Nine nerve sections from unilateral vocal fold paralysis (UVP) patients and seven control nerve sections were analyzed for morphometric parameters including fascicular area, fiber count, fiber density, fiber packing, mean g-ratio, and fiber diameter distribution. Nerves from UVP patients were also compared as a function of time since UVP onset. RESULTS: In comparison to control nerves, paralyzed nerves were found to have significantly lower fiber densities and fiber packing, higher mean g-ratio values, and a shift in diameter distributions toward smaller diameter fibers. With respect to paralysis duration, no significant differences were observed except in fiber diameter distributions, where those with paralysis for >2 years had distributions that were significantly shifted toward smaller diameter fibers. CONCLUSIONS: The morphometric data presented here suggest that correlations between the time since onset of vocal fold paralysis and reinnervation outcomes may be due to fiber size changes in the paralyzed nerve over time.

Unilateral Vocal Fold Immobility in Children.

Unilateral vocal fold paralysis (UVFP) in children may cause dysfunction in voice, swallowing, and breathing, thus influencing all components of laryngeal function. UVFP in children is usually the result of iatrogenic injury. The approach to patients with suspected UVFP should involve a detailed patient history, a focused physical examination with flexible nasopharyngoscopy, and relevant imaging if the cause of UVFP is uncertain. Management aims to strengthen voice, decrease aspiration, and improve quality of life. Laryngeal reinnervation is becoming more common, potentially offering permanent improvement in vocal and swallowing function through increasing bulk and tone to the paralyzed vocal fold.

Nebulizer Use in Adults With Subglottic Stenosis: A Survey Study.

OBJECTIVE:: People with subglottic stenosis (SGS) may experience laryngeal symptoms that pose significant challenges to management. This study surveyed the use and effects of nebulized treatments on laryngeal symptoms in a large cohort of adults with SGS. METHODS:: A survey of nebulized treatment practices was distributed to an online international community of over 2000 members; 316 individuals (298 female, 1 male, 17 declined to specify; age 25+ years) completed the survey. RESULTS:: Nearly half (144) of participants had tried a nebulizer in the past. Among those, half currently used nebulized treatments; the majority used these treatments regularly and for greater than 1 year. The most frequently reported treatments included isotonic saline (0.9% Na+Cl-) or hypertonic saline (3% Na+Cl-). Symptoms improved by these treatments included thick mucus (69%), cough (57%), throat dryness (31%), stridor (28%), and voice (15%). The most frequently reported limitations to nebulizer use included time, noise, and portability. CONCLUSION:: Among those individuals with SGS who have tried nebulized treatments, nearly half experienced relief from secondary laryngeal symptoms. Additional efficacy research is needed, particularly with respect to treatment type and dosage. The results are encouraging given the adverse impact these laryngeal symptoms can have on activities of daily living and quality of life.

Age at diagnosis, but not HPV type, is strongly associated with clinical course in recurrent respiratory papillomatosis.

BACKGROUND: Recurrent Respiratory Papillomatosis (RRP) is a rare disease characterized by the growth of papillomas in the airway and especially the larynx. The clinical course is highly variable among individuals and there is poor understanding of the factors that drive an aggressive vs an indolent course. METHODS: A convenience cohort of 339 affected subjects with papillomas positive for only HPV6 or HPV11 and clinical course data available for 1 year or more, from a large multicenter international study were included. Exploratory data analysis was conducted followed by inferential analyses with frequentist and Bayesian statistics. RESULTS: We examined 339 subjects: 82% were diagnosed prior to the age of 18 years, 65% were infected with HPV6, and 69% had an aggressive clinical course. When comparing age at diagnosis with clinical course, the probability of aggressiveness is high for children under five years of age then drops rapidly. For patients diagnosed after the age of 10 years, an indolent course is more common. After accounting for confounding between HPV11 and young age, HPV type was minimally associated with aggressiveness. Fast and Frugal Trees (FFTs) were utilized to determine which algorithms yield the highest accuracy to classify patients as having an indolent or aggressive clinical course and consistently created a branch for diagnostic age at ~5 years old. There was no reliable strong association between clinical course and socioeconomic or parental factors. CONCLUSION: In the largest cohort of its type, we have identified a critical age at diagnosis which demarcates a more aggressive from less aggressive clinical course.