Tracheostomy care quality improvement in low- and middle-income countries: A scoping review.

Tracheostomy is a lifesaving, essential procedure performed for airway obstruction in the case of head and neck cancers, prolonged ventilator use, and for long-term pulmonary care. While successful quality improvement interventions in high-income countries such as through the Global Tracheostomy Collaborative significantly reduced length of hospital stay and decreased levels of anxiety among patients, limited literature exists regarding tracheostomy care and practices in low and middle-income countries (LMIC), where most of the world resides. Given limited literature, this scoping review aims to summarize published tracheostomy studies in LMICs and highlight areas in need of quality improvement and clinical research efforts. Based on the PRISMA guidelines, a scoping review of the literature was performed through MEDLINE/PubMed and Embase using terms related to tracheostomy, educational and quality improvement interventions, and LMICs. Publications from 2000-2022 in English were included. Eighteen publications representing 10 countries were included in the final analysis. Seven studies described baseline needs assessments, 3 development of training programs for caregivers, 6 trialed home-based or hospital-based interventions, and finally 2 articles discussed development of standardized protocols. Overall, studies highlighted the unique challenges to tracheostomy care in LMICs including language, literacy barriers, resource availability (running water and electricity in patient homes), and health system access (financial costs of travel and follow-up). There is currently limited published literature on tracheostomy quality improvement and care in LMICs. Opportunities to improve quality of care include increased efforts to measure complications and outcomes, implementing evidence-based interventions tailored to LMIC settings, and using an implementation science framework to study tracheostomy care in LMICs.

Comparative Study of Pharyngeal Flap Outcomes between Children With 22q11.2 Deletion Syndrome and Non-Syndromic Cleft Lip and Palate.

BACKGROUND: Management of velopharyngeal insufficiency (VPI) in 22q11.2 deletion syndrome (22q) is challenging. This study compares pharyngeal flap outcomes in children with 22q to those with non-syndromic cleft lip and palate (CLP) to assess risk of poor speech outcomes and negative sequelae. METHODS: Children with 22q or CLP treated with pharyngeal flap through a multidisciplinary VPI clinic between 2009 and 2020 were retrospectively reviewed. Pre- and postoperative speech assessments, perioperative characteristics, and complications were identified. RESULTS: 36 children with ​22q and 40 with CLP were included. Age at surgery (p=0.121), pre-operative velopharyngeal competence score (VPC) (p=0.702), and pre-operative resonance (p=0.999) were similar between groups. Pharyngeal flaps were wider (p=0.038*) and length of stay longer in the 22q group (p=0.031*). On short term follow 4 months after surgery, similar speech outcomes were seen between groups. At long term follow up >12 months after surgery, 86.7% 22q v. 100% CLP (p=0.122) had improvement in velopharyngeal function, however fewer children with 22q (60.0%) achieved a completely "competent" VPC score compared to those with CLP (92.6%) (p=0.016*). Nasal regurgitation improved for both groups, with a greater improvement in those with 22q (p=0.026*). Revision rate (p=0.609) and new onset OSA (0.999) were similar between groups. CONCLUSION: Children with 22q have improved speech after pharyngeal flap, but may be less likely to reach normal velopharyngeal function over the long term than those with CLP; however, negative sequelae do not differ. Improvement in nasal regurgitation is a uniquely positive outcome in this population.

Barriers to Voice Therapy in Dysphonic Children.

BACKGROUND: The most common etiologies of dysphonia in the pediatric population are vocal fold nodules and muscle tension dysphonia. Vocal therapy is the first line treatment for these disorders in children. Despite this, not all children undergo therapy. The goal of this study is to examine how factors such as patient demographics and parental perceptions differ between children that choose to undergo or not to undergo voice therapy. METHODS: A retrospective review was conducted of all pediatric patients seen at a tertiary voice clinic between January 2014 and December 2017. Patients were included if diagnosed with vocal fold nodules and/or muscle tension dysphonia. Patients were divided into groups of children that received voice therapy at our institution and those that did not. Data include demographics, Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) scores and pediatric Voice Handicap Index (pVHI) scores. Distance to therapy site was approximated using patient zip codes. RESULTS: Three hundred and forty-six children were included, 224 (65%) boys and 122 (35%) girls. In the 2 years following initial diagnosis, 74 (21%) children participated in voice therapy at our institution. Patients who underwent voice therapy were older than those who did not (mean age: 9.1 [SD 3.5] vs 7.6 [SD 3.8] years; P = 0.004). Patients who received voice therapy were more likely to live closer to the therapy site (mean distance: 15.5 [SD 13.0] vs 24.3 [SD 23.9] miles; P< 0.001). Likelihood of receiving voice therapy did not differ by gender or health insurance status (private vs public). Patients who underwent voice therapy had significantly greater CAPE-V Overall Severity scores than those who did not (mean score: 44.6 [SD 19.4] vs 37.4 [SD 18.0]; P = 0.003). Higher CAPE-V Strain scores were associated with increased likelihood of voice therapy. pVHI scores did not differ between the two groups. CONCLUSION: Older age, shorter distance to therapy site, and increased CAPE-V Overall Severity and Strain scores were associated with higher likelihood of receiving voice therapy. Gender, insurance status, and pVHI scores did not affect likelihood of receiving voice therapy. Patients may primarily consider ease of access and necessity of treatment when considering voice therapy.

Videofluoroscopy Versus Nasopharyngoscopy of Lateral Pharyngeal Wall Movement for Assessment of Velopharyngeal Insufficiency.

BACKGROUND: Competent speech requires closure of the velopharyngeal sphincter by dynamic apposition of the velum and posterior and lateral pharyngeal walls. An accurate estimation of lateral pharyngeal wall motion is an important determinant in the planning and the outcome of any operation to correct velopharyngeal insufficiency (VPI). The purpose was to compare the assessment of lateral pharyngeal wall movement by videofluoroscopy (VP) versus nasopharyngoscopy (NP). METHODS: The authors retrospectively reviewed the charts of 269 consecutive patients in our cleft lip/palate clinic from 1982 to 2008 and culled those treated with a pharyngeal flap for VPI. The authors included patients who were evaluated preoperatively by both VP and NP, and had studies of suitable quality. Percentage of lateral pharyngeal wall motion was estimated with each technique and compared for each patient. RESULTS: The authors identified 25 patients who underwent both VP and NP at the same median age (4.7 years). The estimated percentage of lateral pharyngeal wall motion between the 2 techniques was significantly different ( P <0.001). Average lateral pharyngeal wall motion was estimated to be 59±25% (range: 5%-90%) by VP and only 40%±25% (range: 0%-95%) during NP. CONCLUSIONS: VP and NP are complementary, but assessment of lateral pharyngeal wall motion can vary between the 2 methods. The surgeon should be aware of the difference in estimated lateral pharyngeal wall movement when planning a procedure to correct VPI.

Laryngeal pathologies in dysphonic children with Down Syndrome.

INTRODUCTION: Down syndrome is the most common chromosomal abnormality and is associated with a higher incidence of congenital heart defects, which often require surgery within the first year of life. Previous studies have found that children with Down syndrome are at higher risk for subglottic stenosis, vocal fold paralysis, and laryngomalacia. The goal of this study is to review children with Down syndrome presenting with dysphonia and to characterize their laryngeal pathologies. METHODS: A retrospective review was performed of patients with Down syndrome seen at a tertiary pediatric hospital's department of otolaryngology from Jan. 2007-Jul. 2021 for voice-related concerns. Inclusion criteria included age less than 18 years, diagnosis of Trisomy 21, and complaint of dysphonia. The data extracted included history of dysphonia, co-morbidities, demographic information, age at presentation, perceptual voice assessments, voice quality of life scores, acoustic data, laryngoscopic and/or videostroboscopic exams, and surgical procedures. RESULTS: Twenty-three total patients met the study criteria. Of these children, 13 (57%) were male and 10 (43%) were female. The mean age at first presentation was 4.08 years (range 12 days-16.3 years). Eleven of the 23 patients presented within the first 12 months of life. Sixteen patients were diagnosed with vocal fold immobility, 13 of which were left-sided unilateral immobility and the remaining 3 were bilateral immobility. 5 patients were diagnosed with vocal fold nodules. 12 children in the immobility group had a history of cardiothoracic surgery at our institution. Only 3 patients had Consensus Auditory-Perceptual Evaluation of Voice (CAPE-V) assessments, though all three showed overall dysphonia ratings of severely deviant, with roughness and strain scores being the most severe. DISCUSSION: The most common etiology of dysphonia in our Down syndrome patient population was vocal fold immobility and hypomobility, as opposed to vocal fold nodules (which is the most common in the general pediatric population). The higher likelihood of cardiac surgery in patients with Trisomy 21 may result in the increased incidence of vocal fold immobility. There should be a low threshold to refer dysphonic patients with Down syndrome for laryngoscopic evaluation, as treatment options may be available.

Laryngeal Web in the Pediatric Population: Evaluation and Management.

OBJECTIVE: To discuss the presentation, evaluation, and management of pediatric laryngeal web. STUDY DESIGN: Retrospective case series. SETTING: Single tertiary care center. SUBJECTS: All patients with laryngeal web at Boston Children's Hospital in the past 22 years. METHODS: No exclusion criteria. Charts mined for age at presentation, presenting symptoms, degree/location of web, associated syndromes, number/type of surgical procedures, and postoperative outcomes. RESULTS: Thirty-seven patients were included (13 male, 24 female). Average age at diagnosis was 3.7 years (0-19.5 years). Mean follow-up was 4.4 years (range, 0-16.4 years). There were 26 congenital webs (70.2%) and 11 acquired webs (29.8%). Presenting symptoms were vocal (29 patients, 78.4%) and respiratory (22 patients, 60%). Underlying syndromes or synchronous airway lesions included the following: premature (n = 5), congenital heart disease (n = 18), subglottic stenosis (n = 5), 22q11.2 deletion syndrome (n = 10), and recurrent respiratory papillomatosis (n = 4). There were 20 type 1 webs, 6 type 2 webs, 8 type 3 webs, and 3 type 4 webs; 10 had subglottic extension of the laryngeal web. Twelve patients were managed conservatively with observation. Eighty-four interventions were performed: 18 open and 66 endoscopic (sharp division, 32; dilation, 33; mitomycin C, 14; laser, 5; keel, 6; triamcinolone injection, 8; stent, 15; removal of granulation tissue, 5). Tracheotomy was required in 11 patients, and 5 patients were decannulated. Voice improved in 12 patients, with respiratory symptoms in 12 patients. Web recurred in 17 patients. One patient died due to airway complications. CONCLUSIONS: Pediatric laryngeal web is an uncommon but challenging lesion. Patients need to be evaluated for comorbid syndromes and synchronous airway lesions. Management includes open and endoscopic procedures. Procedures should be tailored to the child's presentation.

A retrospective review of the progression of pediatric vocal fold nodules.

IMPORTANCE: To our knowledge, the rate of change in the size of pediatric vocal fold nodules (VFNs) has not been investigated. Improved understanding of the factors that affect change in VFN size may help to better guide treatment decisions and counselling of families. OBJECTIVE: To characterize the rate of change in the size of pediatric VFNs over time and to identify which factors affect increased rates of improvement. DESIGN, SETTING, AND PARTICIPANTS: Retrospective review of 67 children evaluated in a voice clinic between 2002 and 2011 with a primary diagnosis of VFNs. EXPOSURE: No treatment or behavioral modification only (n = 19) vs targeted voice therapy with or without the treatment of associated conditions (gastroesophageal reflux and allergic rhinitis) (n = 45) vs surgical intervention (n = 3). MAIN OUTCOMES AND MEASURES: Change in VFN grade (graded according to a previously validated scale based on size) over time. RESULTS: Sixty-seven patients with a median (range) age of 6.0 (3.8-20.6) years were analyzed. Median (range) follow-up was 25 (1-119) months. The rate of change in VFN grade over time was significantly associated with large baseline VFN size (P < .001) and targeted voice therapy with or without the management of associated conditions or surgery (P = .01); the association with postpubescent age was not significant (P = .09). The rate of change in VFN grade was not significantly different at 1 and 3 years postbaseline (P = .33). CONCLUSIONS AND RELEVANCE: Baseline VFN size, treatment, and patient age are important in predicting the rate of improvement in nodule size over time. Rate of change in VFN size is a gradual decrease that is steady over time. This information can be used to help guide treatment decisions and counsel families of children with VFNs regarding expectations for improvement. Additional study is needed to evaluate whether the same factors that influence nodule size similarly influence parental perception of voice and expert perceptual voice analysis.

Validation of a pediatric vocal fold nodule rating scale based on digital video images.

OBJECTIVES: We sought to create a validated scale of vocal fold nodules in children, based on digital video clips obtained during diagnostic fiberoptic laryngoscopy. METHODS: We developed a 4-point grading scale of vocal fold nodules in children, based upon short digital video clips. A tutorial for use of the scale, including schematic drawings of nodules, static images, and 10-second video clips, was presented to 36 clinicians with various levels of experience. The clinicians then reviewed 40 short digital video samples from pediatric patients evaluated in a voice clinic and rated the nodule size. Statistical analysis of the ratings provided inter-rater reliability scores. RESULTS: Thirty-six clinicians with various levels of experience rated a total of 40 short video clips. The ratings of experienced raters (14 pediatric otolaryngology attending physicians and pediatric otolaryngology fellows) were compared with those of inexperienced raters (22 nurses, medical students, otolaryngology residents, physician assistants, and pediatric speech-language pathologists). The overall intraclass correlation coefficient for the ratings of nodule size was quite good (0.62; 95% confidence interval, 0.52 to 0.74). The p value for experienced raters versus inexperienced raters was 0.1345, indicating no statistically significant difference in the ratings by these two groups. The intraclass correlation coefficient for intra-rater reliability was very high (0.89). CONCLUSIONS: The use of a dynamic scale of pediatric vocal fold nodule size most realistically represents the clinical assessment of nodules during an office visit. The results of this study show a high level of agreement between experienced and inexperienced raters. This scale can be used with a high level of reliability by clinicians with various levels of experience. A validated grading scale will help to assess long-term outcomes of pediatric patients with vocal fold nodules.

Predictors of velopharyngeal insufficiency after Le Fort I maxillary advancement in patients with cleft palate.

PURPOSE: Approximately 25% to 40% of patients with cleft lip/palate develop maxillary retrusion that requires Le Fort I osteotomy. Maxillary advancement brings the soft palate forward, and this may cause velopharyngeal insufficiency (VPI). The goal of this study was to identify predictors that place patients with repaired cleft palate at risk of developing VPI after Le Fort I advancement. MATERIALS AND METHODS: This was a retrospective study of nonsyndromic patients with cleft lip/palate who had a Le Fort I osteotomy between 2000 and 2008. Charts were reviewed and data were collected on patient characteristics, preoperative speech assessments, and nasopharyngoscopic reports. Pre- and postoperative cephalometric radiographs were used to measure maxillary advancement and to assess the structure of the velopharynx. Simple logistic regression analysis examined the association between each predictive variable and postoperative VPI, as indicated by need for pharyngeal flap. Predictors with P ≤ .10 were included in the multivariate regression model. In both the univariate and the multivariate analyses, P ≤ .05 was considered statistically significant. RESULTS: Univariate analysis showed a significant association between preoperative soft palatal length and need for a pharyngeal flap (P = .005). By multivariate analysis, both preoperative soft palatal length and postoperative pharyngeal depth were associated with need for pharyngeal flap (P = .003 and P = .030). CONCLUSION: This study shows that a short soft palate is associated with VPI after Le Fort I osteotomy. Assessment of palatal length and pharyngeal depth on cephalometric radiographs is helpful in predicting postoperative VPI and need for a pharyngeal flap in patients with cleft palate after maxillary advancement.

Minimally invasive endoscopic management of subglottic stenosis in children: success and failure.

OBJECTIVE: To assess the efficacy and safety of endoscopic management of subglottic stenosis both as a primary and as an adjunctive treatment in the pediatric population. METHODS: Retrospective review of pediatric patients with subglottic stenosis undergoing endoscopic airway procedures at a tertiary care pediatric medical center. Outcomes were assessed by systematic review to determine the success and failure of the endoscopic approach. RESULTS: Forty patients (22 male, 18 female) underwent endoscopic interventions for a diagnosis of subglottic airway stenosis between 2003 and 2006. Age ranged from 22 days old to 20 years old. Recorded degree of subglottic stenosis ranged from 10% to 99%. Fifty-three percent (21/40) had a history of prematurity, and 40% (16/40) had secondary airway diagnoses. Twenty-four patients underwent an endoscopic intervention initially (including laser or dilation, with or without topical mitomycin treatment), including four patients who underwent tracheostomy prior to the first endoscopic intervention. Sixteen underwent laryngotracheoplasty initially, including ten patients who underwent tracheostomy prior to the laryngotracheoplasty. Endoscopic treatment resulted in resolution of symptoms, and/or decannulation, and no further need for an open procedure in 58% of patients. Of the 24 patients undergoing endoscopic interventions initially, 14 patients underwent two or more endoscopic interventions, and 10 patients subsequently required tracheostomy or laryngotracheoplasty. When endoscopic procedures were used as an adjunct to laryngotracheoplasty, 60% (12/20) had resolution of symptoms, underwent decannulation, and did not require tracheostomy or revision laryngotracheoplasty. CONCLUSIONS: The endoscopic approach can be successful in the management of properly selected patients with subglottic stenosis, either as the initial treatment modality or as an adjunctive treatment in cases of re-stenosis after open airway surgery. The likelihood of success with a minimally invasive procedure as the primary treatment decreases with worsening initial grade of subglottic stenosis.

A grading scale for pediatric vocal fold nodules.

OBJECTIVE: To design a grading scale for vocal fold nodules in pediatric patients. STUDY DESIGN AND SETTING: We conducted a prospective study in which a grading scale for vocal nodule size and contour based on static fiberoptic images of pediatric larynges was developed to achieve the scale presented here. RESULTS: Twenty-eight health care professionals each rated 28 images of pediatric vocal fold nodules. The intraclass correlation for nodule size was strong (0.77; 95% confidence interval, 0.67-0.87). The kappa statistic for nodule contour was mild (0.35; 95% confidence interval, 0.33-0.37). Agreement between experienced and other raters found no significant difference for the nodule size or contour grade of a given image. CONCLUSIONS: A grading scale for pediatric vocal fold nodules is presented. Interrater reliability for nodule size is high and can be reliably used by health care professionals with varying levels of experience. SIGNIFICANCE: A validated grading scale facilitates objective analysis of outcomes when studying and following patients with vocal nodules.

The presentation and management of laryngeal cleft: a 10-year experience.

OBJECTIVE: To review the presentation and associated congenital abnormalities of laryngeal cleft and present guidelines for its evaluation and management. DESIGN: A 10-year retrospective study (1994-2004) with institutional review board approval. SETTING: Two pediatric tertiary care medical centers. PATIENTS: Twenty-two pediatric patients (mean age, 21 months) with laryngeal cleft. INTERVENTION: Surgical repair of laryngeal cleft. MAIN OUTCOME MEASURES: Sex, age, symptoms, other associated abnormalities, method of evaluation, type of laryngeal cleft, method of surgical repair, treatment outcome, complications, and long-term follow-up. RESULTS: All 22 patients underwent surgical repair for laryngeal cleft. Airway endoscopy confirmed the following types of laryngeal clefts: type 1 (n = 3), type 2 (n = 10), and type 3 (n = 9). Surgical repair techniques included an open approach with or without interposition graft (n = 16) and an endoscopic approach (n = 6). CONCLUSIONS: Early diagnosis and proper repair of laryngeal cleft are essential to prevent pulmonary damage and associated morbidity. Each patient should be assessed properly, and the surgical approach should be individualized based on the symptoms, other associated findings on airway endoscopy, and type of cleft.

Perceptual voice characteristics in pediatric unilateral vocal fold paralysis.

OBJECTIVE: To describe the voice characteristics of pediatric unilateral vocal fold paralysis (UVFP). STUDY DESIGN AND SETTING: Retrospective series from a children's hospital, 1996 to present. RESULTS: Forty-two patients with a mean age of 7.1 years were diagnosed with UVFP in our voice clinic (prevalence = 5.4%). Paralysis was left sided in 88%, and was most commonly seen after cardiac surgery (28.5%) and prolonged intubation/prematurity (16.7%). Voice analysis showed a moderate degree of breathiness, mild-to-moderate hoarseness and straining, mild muscle tension, and soft loudness. Twenty-six percent of patients required surgical intervention, including injection into the paralyzed fold (7 patients) and medialization thyroplasty (4 patients). Pre-operatively, breathiness was worse (P < .05) in patients undergoing surgical intervention. CONCLUSIONS: Voice characteristics of pediatric UVFP include increased breathiness, hoarseness, straining, muscle tension, and soft loudness. One-fourth of patients underwent surgical intervention; breathiness was the predominant abnormal voice characteristic in the operative cohort. SIGNIFICANCE: The voice characteristics of pediatric patients with UVFP are described. EBM RATING: C-4.

The biology and management of laryngeal neurofibroma.

OBJECTIVES: To review the presentation of laryngeal neurofibroma, including its association with neurofibromatosis types 1 and 2, and present guidelines for its management. DESIGN: Retrospective study. PATIENTS: Five pediatric patients with laryngeal neurofibroma, 4 girls (80%) and 1 boy (20%), were treated at a tertiary pediatric medical center from 1973 through 2003. Recorded data included age at initial presentation, sex, symptoms, significant medical and family history, preoperative evaluation, location of the tumor, surgical procedure, complications, outcome, and recurrence. RESULTS: The 5 patients presented with stridor and cafe-au-lait spots at or shortly after birth. All patients were diagnosed as having neurofibromatosis type 1 by the established criteria. Studies evaluating the disease processes included plain radiography, computerized tomography, magnetic resonance imaging, barium swallow, and laryngoscopy and bronchoscopy under anesthesia. Pathologic examination of biopsy specimens from all patients showed neurofibromas with plexiform and/or diffuse features. Treatments included tracheotomy (n = 4), carbon dioxide laser excision (n = 4), modified neck dissection (n = 3), partial pharyngectomy (n = 1), supraglottic laryngectomy (n = 1), and endoscopic hemilaryngectomy (n = 1). Three patients were successfully decannulated. Follow-up ranged from 1 to 15 years. One patient was lost to follow-up. No evidence of malignant degeneration was noted. CONCLUSIONS: Neurofibroma of the larynx is a rare condition that should be considered in the differential diagnosis of children with a submucosal laryngeal mass. In our series, all patients had associated neurofibromatosis type 1. Complete surgical excision is the treatment of choice in cases of localized small lesions. To prevent debilitating outcomes due to aggressive surgery, minimally invasive procedures (partial excision via endoscopic approach) may be preferable for larger lesions that infiltrate the surrounding vital structures. Long-term follow up of these patients is essential owing to the possibility of malignant transformation.

The role of mitomycin in the prevention and treatment of scar formation in the pediatric aerodigestive tract: friend or foe?

OBJECTIVE: To evaluate the role of mitomycin in the prevention and treatment of scar formation in the pediatric aerodigestive tract. DESIGN: Prospective study; institutional review board-approved clinical trial. SETTING: Tertiary care pediatric medical center. PATIENTS: Fifteen patients; choanal atresia in 5 patients, airway stenosis in 8 patients, hypopharyngeal stenosis in 1 patient, and esophageal stenosis in 1 patient. OUTCOME: The efficacy and safety of mitomycin in the prevention of scar formation. INTERVENTION: All patients underwent surgical repair of the stenotic area, followed by topical application of mitomycin (1 mL of 0.4 mg/mL) for 4 minutes. RESULTS: Ten patients (67%) showed major improvement, 4 patients (27%) showed minor improvement, and 1 patient (7%) showed no improvement. CONCLUSION: Topical application of mitomycin can play an effective role in the prevention and treatment of scar formation in the aerodigestive tract.