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The upper aerodigestive system is closely intertwined from an embryologic and functional perspective. Laryngotracheal anatomic abnormalities, such as laryngomalacia, stenosis, vocal cord paralysis, and laryngeal clefts, affect not only the respiratory function but also the swallow function. Laryngotracheal pathology can interfere with the suck-swallow-breathe mechanism in infants. It can also exacerbate gastroesophageal reflux. Chronic aspiration secondary to laryngotracheal anomalies can result in respiratory and pulmonary complications. Surgical treatment of laryngotracheal anomalies can also cause transient or long-term swallow dysfunction. Multidisciplinary approaches and clinical assessment of swallowing are important in patients with laryngotracheal pathology., Competing Interests: Disclosure The authors have no financial disclosures., (Copyright © 2024 Elsevier Inc. All rights reserved.)

Background: Ohtahara syndrome is a progressive developmental and epileptic encephalopathy that manifests in the early infantile period. This rare condition is characterized by intractable seizures, psychomotor retardation, and poor prognosis. To date, there are a handful of case reports regarding the anesthetic management of children with Ohtahara syndrome. However, limited reports exist of patients with Ohtahara syndrome who present with difficult airways. This report describes our airway findings and general anesthetic management of a pediatric patient with Ohtahara syndrome undergoing diagnostic bronchoscopy for severe inspiratory stridor., Case Presentation: A 14-month-old, 9 kg, male patient with Ohtahara syndrome presented with a year-long history of severe inspiratory stridor and was scheduled for bronchoscopy with lavage. On exam, the patient had noisy breathing, was non-verbal with developmental delay, and had poor head control with significant central hypotonia. The patient was induced with ketamine and general anesthesia was maintained with propofol. Bronchoscopic evaluation was completed uneventfully and revealed a diagnosis of laryngotracheomalacia. The patient's breathing was maintained spontaneously throughout the procedure and no seizures were noted. In the post anesthesia care unit, the patient's respiratory and cardiovascular function were stable., Conclusions: This report documents the unusual finding of severe inspiratory stridor in a 14-month-old child diagnosed with Ohtahara syndrome and our anesthetic management during their diagnostic bronchoscopy. Currently, documentation of complex airway pathology present in patients with Ohtahara syndrome is limited and should be further evaluated. This will assist pediatric anesthesiologists as these patients may require careful preoperative assessment, thoughtful airway management, and surgical alternatives on standby., (© 2024. The Author(s).)

Background: Moderate laryngomalacia lies in the grey zone where the parents and treating team might hesitate to decide the treatment plan for the child. Neurodevelopmental assessment of the child was neither assessed nor incorporated in surgical decision-making in the past., Objectives: To evaluate the neurodevelopmental outcome of moderate laryngomalacia treatment strategies (surgery versus conservative)., Methods: We conducted an observational multicentric cohort study. We compared two groups of patients according to their moderate laryngomalacia treatment strategy: surgical treatment and no treatment. The neurodevelopmental outcome was assessed by Griffiths-III developmental scales., Results: A total of 150 children were diagnosed with moderate laryngomalacia of which 56 were successfully treated with surgery and 94 improved without intervention. The median Griffiths-III developmental quotients (DQs) of subscales A, B, C, E, and general development were significantly higher in the surgically treated group compared to conservatively treated ones., Conclusion: Untreated moderate laryngomalacia cases have worse neurodevelopmental outcomes than surgically treated cases., Competing Interests: Declaration of competing interest There is no conflict of interest., (Copyright © 2024 Elsevier B.V. All rights reserved.)

Purpose: This study aimed to investigate the role of nap polysomnography (NPSG) in predicting treatment strategies for infants with moderate to severe laryngomalacia and to explore the association between obstructive sleep apnea (OSA) severity, weight gain, and laryngomalacia severity., Methods: A retrospective analysis was conducted on infants diagnosed with moderate to severe laryngomalacia who underwent NPSG between January 2019 and June 2023. Clinical variables, NPSG parameters, and treatment decisions were collected. Weight gain rate and its correlation with NPSG indices were assessed. Logistic regression analyses were performed to predict treatment strategies based on NPSG findings., Results: Of the 39 infants included (median age: 3.3 months), 77% exhibited OSA, with 69% having moderate to severe OSA [apnea-hypopnea index (AHI) > 5/h]. Weight gain rate correlated negatively with indices of OSA severity, including the hypopnea index (HI) and the AHI. In a multiple logistic regression analysis incorporating the severity of OSA (AHI), weight gain rate, and laryngomalacia severity, only AHI predicted the decision for surgical or non-invasive ventilation treatment (OR = 2.1, CI 95 [1.6; 2.8], p ≤ 10 -4 ). The weight gain rate was predicted (r 2  = 0.28) by the AHI and the presence of retractions of auxiliary inspiratory muscles., Conclusion: This study underscores the importance of NPSG in assessing infants with moderate to severe laryngomalacia. The AHI from NPSG emerged as a potential predictor for treatment decisions and weight gain rate, emphasizing its clinical relevance. These findings advocate incorporating NPSG into the diagnostic and management process for infants with laryngomalacia., (© 2024. The Author(s).)

Obstructive sleep apnea in newborns and infants presents a unique challenge with distinct differences in sleep physiology, etiologies, and management compared to older children. The indications for and interpretation of polysomnography are less well defined in infants. There are also no broadly accepted clinical practice guidelines for treating sleep apnea in this age group. Etiologies include general causes of upper airway obstruction in infants such as laryngomalacia, micrognathia, and nasal obstruction in addition to adenotonsillar hypertrophy. Treatment strategies must be tailored to the specific anatomic features and comorbidities of the specific patients and often require a multidisciplinary approach., Competing Interests: Disclosure The authors have nothing to disclose., (Copyright © 2024 Elsevier Inc. All rights reserved.)

Background: Diagnosis and treatment of obstructive sleep apnea (OSA) in infants and young children is challenging because of its clinical heterogeneity and lack of age-specific guidelines., Aim: We report the management and treatment outcome of OSA in children below 2 years of age. Treatment decisions were based upon the pattern of upper airway (UA) obstruction, clinical presentation and OSA severity., Methods: Retrospective, non-randomized observational cohort study at a tertiary center. Children with OSA who underwent an UA evaluation (drug-induced sleep endoscopy or direct laryngoscopy) were included., Results: We studied 100 patients, 57 boys and 43 girls, age 0.72 years (0.0-2.0) and OSA confirmed by polysomnography. Multilevel UA collapse was present in 26%, (adeno)tonsillar hypertrophy in 31% and 21% had laryngomalacia. Laryngomalacia was more common in children below 6 months of age and adenotonsillar hypertrophy was observed mainly in children >1.5 year of age. Surgical and nonsurgical treatment guided by UA findings, improved OSA severity at group level with a significant reduction (p < 0.001) in obstructive apnea/hypopnea index from 10.8/h (2.1-99.1) to 1.7/h (0.0-73.0), an improvement in mean oxygen saturation from 96.9% (88.9-98.4) to 97.4% (92.3-99.0), in minimal oxygen saturation from 85.4% (37.0-96.0) to 88.8% (51.0-95.5) and oxygen desaturation index from 5.1/h (0.2-52.0) to 1.3/h (0.0-47.8)., Conclusion: Multidisciplinary management of young children with OSA guided by the pattern of UA obstruction and OSA severity, reduces OSA severity. The pattern of UA obstruction changes in the first 2 years of life from a dynamic collapse to structural abnormalities., (© 2024 Wiley Periodicals LLC.)

Stridor in an infant is a significant clinical sign; the primary objectives are to ensure that the airway is safe and to arrange timely, appropriate management. A structured history, examination and targeted investigations will determine the cause and guide care.Laryngomalacia is the most common cause of stridor in an infant. The stridor tends to start shortly after birth, classically presenting as a positional stridor in the first month, which gradually resolves before 12-18 months of age in mild cases. There is a wide spectrum of severity; few require surgical intervention. This article will outline how the infant is appropriately assessed and managed., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2024. No commercial re-use. See rights and permissions. Published by BMJ.)

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., Competing Interests: Declaration of competing interest none., (Copyright © 2024 Elsevier B.V. All rights reserved.)

Objective: To compare supraglottoplasty versus non-surgical treatment in children with laryngomalacia and mild, moderate and severe obstructive sleep apnoea., Methods: Patients were classified based on their obstructive apnoea hypopnoea index on initial polysomnogram, which was compared to their post-treatment polysomnogram., Results: Eighteen patients underwent supraglottoplasty, and 12 patients had non-surgical treatment. The average obstructive apnoea hypopnoea index after supraglottoplasty fell by 12.68 events per hour ( p = 0.0039) in the supraglottoplasty group and 3.3 events per hour ( p = 0.3) in the non-surgical treatment group. Comparison of the change in obstructive apnoea hypopnoea index in the surgical versus non-surgical groups did not meet statistical significance ( p = 0.09)., Conclusion: All patients with laryngomalacia and obstructive sleep apnoea had a statistically significant improvement in obstructive apnoea hypopnoea index after supraglottoplasty irrespective of obstructive sleep apnoea severity, whereas patients who received non-surgical treatment had more variable and unpredictable results. Direct comparison of the change between the two groups did not find supraglottoplasty to be superior to non-surgical treatment. Larger prospective studies are recommended.

Objective: This review sought to determine the characteristics of adults diagnosed with new onset laryngomalacia including airway symptoms, laryngoscopic findings, treatments, and outcomes. Moreover, we wanted to highlight suspected limitations in the literature., Data Sources: Studies were identified through CINAHL, Cochrane Review, PubMed, and Scopus published between 1966 and 2023., Review Methods: The search was performed in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-analysis Extension for Scoping Reviews checklist by 2 independent investigators. A meta-analysis of proportions and continuous measures was conducted., Results: Of the 1121 abstracts identified, 33 articles pertaining to laryngomalacia in the adult population were included. The most common presenting symptoms were stridor at rest (78.3%, 65.1-88.3) and dyspnea with exertion (83.8%, 64.8-96.3). The most suspected etiology was exercise-induced (86.0%, 69.4-95.5), and the most common description of laryngomalacia on visualization was collapse of supraglottic structures during exercise (93.3%, 79.0-99.1). Nonsurgical options were attempted in 87.0% (54.0-99.1), which included oral appliances, respiratory retraining, breathing techniques, and working with a speech pathologist. Surgical options were ultimately performed in 84.2% (75.0-91.0). Complete resolution of symptoms following therapy was seen in 61.9% (48.0-74.6)., Conclusion: Adult onset laryngomalacia is difficult to characterize. It typically presents in patients during exercise, with neurological injury, or idiopathically. Surgical management can lead to improvement or complete resolution of symptoms. The need for a universal nomenclature is highlighted in this review, as it is inconsistently classified., (© 2024 American Academy of Otolaryngology–Head and Neck Surgery Foundation.)

Objective: To examine the prevalence and characteristics of dysphagia and suck-swallow-breath incoordination as phenotypes of oral feeding difficulties., Method: A cross-sectional study with secondary data collected consecutively over 2 years from October 2020 to October 2022 to measure the prevalence of swallowing and oral feeding difficulty in preterm infants using Flexible endoscopic evaluation of swallowing examination at the tertiary Integrated Dysphagia Clinic., Results: The prevalence of swallowing disorders was 25 % and the prevalence of suck-swallow-breath incoordination was 62.5 %. The significant risk factor that may show a possible correlation with oral feeding difficulty was mature post-menstrual age (p = 0.006) and longer length of stay (p = 0.004). The dominant percentage of upper airway abnormality and disorder were retropalatal collapse (40 %), laryngomalacia (42.5 %), paradoxical vocal cord movement (12.5 %), and gastroesophageal reflux disease (60 %). The dominant characteristic of oral motor examination and flexible endoscopic evaluation of swallowing examination was inadequate non-nutritive sucking (45 %), inadequate postural tone (35 %), and inadequate nutritive sucking (65 %)., Conclusion: Dysphagia in preterm infants is mostly observed in those with mature post-menstrual age, longer length of stay, and the presence of gastroesophageal reflux disease with inadequate non-nutritive sucking and nutritive sucking abilities. Suck-swallow-breath incoordination is primarily observed in those with immature post-menstrual age, a higher prevalence of cardiopulmonary comorbidity, and a higher prevalence of upper airway pathologies (laryngomalacia, paradoxical vocal cord movement) with inadequate nutritive sucking ability., Competing Interests: Conflicts of interest The authors declare no conflicts of interest., (Copyright © 2023 Sociedade Brasileira de Pediatria. Published by Elsevier Editora Ltda. All rights reserved.)

Objectives: To analyze characteristics of children treated for laryngomalacia to determine predictive factors and provide an updated meta-analysis on outcomes., Methods: A systematic review was conducted according to PRISMA guidelines from inception to May 2, 2023, using CINAHL, PubMed, and Scopus databases. Study screening, data extraction, quality rating, and risk of bias assessment were performed by 2 independent reviewers. Data were meta-analyzed using fixed-/random-effects model to derive continuous measures (mean), proportions (%), and mean difference (Δ) with 95% confidence interval (CI)., Results: 100 articles were identified with information on outcomes of pediatric patients with laryngomalacia (N = 18,317). The mean age was 10.6 months (range: 0 to 252, 95%CI: 9.6 to 11.6, p = 0.00) with a 1.4:1 male to female ratio. Many patients presented with stridor (87.9%, 95% CI: 69.8 to 98.4), and the most common comorbidity at time of diagnosis was gastroesophageal reflux disease (48.8%, 95%CI: 40.9 to 56.8). Based on the patient population included in our analysis, 86.1% received supraglottoplasty (95% CI: 78.7 to 92.1). A total of 73.6% (95% CI: 65.5 to 81.0) had reported complete resolution of symptoms. For patients with a concurrent diagnosis of sleep disordered breathing receiving supraglottoplasty, the apnea-hypopnea index improved with a mean difference of -10.0 (95%CI: 15.6 to -4.5) events per hour post-treatment., Conclusions: Laryngomalacia continues to be a common problem in the pediatric population. Supraglottoplasty remains an effective treatment option leading to symptomatic improvement in many cases. For those with concurrent sleep disordered breathing, supraglottoplasty lowers the apnea-hypopnea index., Competing Interests: Declaration of competing interest None., (Copyright © 2024 Elsevier B.V. All rights reserved.)

Objective: Postoperative airway concerns persist despite a low rate of post-supraglottoplasty complications for children with laryngomalacia. The objective of this study is to determine the factors associated with the need for intensive care unit (ICU) admission following supraglottoplasty., Methods: A 7-year retrospective cohort analysis was conducted between 2014 and 2021. A patient requiring ICU level of care was defined as the use of respiratory support such as intubation, positive pressure ventilation, high-flow nasal cannula, or multiple doses of nebulized epinephrine., Results: About 134 medical charts were reviewed; 12 patients were excluded because of concurrent surgery. Age at the time of surgery was 2.8 (4.3) months (median [interquartile range]). About 33 (27.0%) ultimately required ICU-level care. Prematurity (odds ratio [OR] 13.8), neurological condition (OR ∞), American Society of Anesthesiology class 3-4 (OR 6.5), and younger age (OR 1.8) were more likely to require ICU admission. No patient above 10 months of age needed ICU monitoring. The use of respiratory support justifying ICU was known within the first 4 h after surgery for almost all (32/33, 97%) of these patients. 4/33 (12.1%) were kept intubated and the remaining needed non-invasive ventilation. Only one patient (1/122, 0.8%) was reintubated 12 h after surgery for progressive respiratory distress., Conclusion: Approximately a quarter of patients required ICU-level care after supraglottoplasty. For nearly all patients without comorbidities requiring ICU, this can be safely predicted within the first 4 h after surgery. Our data suggest that selected patients undergoing supraglottoplasty may be safely monitored outside of an ICU setting after an observation period in the post-anesthesia care unit., Level of Evidence: 4 Laryngoscope, 134:466-470, 2024., (© 2023 The Authors. The Laryngoscope published by Wiley Periodicals LLC on behalf of The American Laryngological, Rhinological and Otological Society, Inc.)

Introduction: Obstructive sleep apnea (OSA) is highly prevalent in infants with laryngomalacia (LM). Previous studies have reported high rates of persistent OSA following supraglottoplasty (SGP). The objective of this study is to describe the findings on drug-induced sleep endoscopy (DISE) in infants with LM., Methods: A retrospective review was performed including infants (12.5 months of age or less) with LM who underwent DISE along with direct laryngoscopy and bronchoscopy (DLB). Data collected included clinical presentation, swallow study results, sleep study results, past medical and surgical history, and postoperative outcomes when follow-up data was available. The findings on DISE as well as DLB were reviewed and described., Results: Thirty-five infants were included in the study with a mean age of 0.42 years (range 0.04-1.04). Obstructive sleep-disordered breathing (oSDB) and/or OSA were identified in 26 patients (74.3%) in addition to 7 patients (20%) with noisy breathing during sleep. DISE demonstrated LM in 31 patients (89%). DISE identified 12 patients (34%) with partial or complete obstruction at the level of the tongue base. DLB identified 14 patients (40%) with a secondary airway abnormality including 7 patients (20%) with type 1 laryngeal cleft and 4 patients (11%) with grade 1 subglottic stenosis. Eighteen patients (51%) underwent SGP., Conclusion: DISE at the time of airway endoscopy for infants with LM can be helpful in identifying additional sites of obstruction including the tongue base. Glossoptosis may explain and/or predict the previously reported persistence of OSA following SGP. Additionally, DISE can complement awake flexible laryngoscopy in the assessment of severity of LM., Competing Interests: Declaration of competing interest The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper., (Copyright © 2023 Elsevier B.V. All rights reserved.)

Objective: To analyze the clinical characteristics of laryngomalacia in Chinese children and explore the surgical efficacy and factors influencing severe laryngomalacia., Methods: Children (0-18 years) diagnosed with laryngomalacia in our hospital from January 2016 to January 2022 were enrolled in this study. Clinical data of patients, including general conditions, clinical symptoms, grading and classification, medical comorbidities, surgical efficacy, and the risk factors influencing severe laryngomalacia were retrospectively analyzed., Results: A total of 1810 children were enrolled (male:female; 2.02:1), among which most were infants under 1 year (77.18%). Inspiratory laryngeal stridor (69.56%) was the most common symptom. Most patients had mild laryngomalacia (79.28%), with type IV laryngomalacia being the most common classification (52.27%). Congenital heart disease (37.85%) was the most common medical comorbidity. A total of 168 severe laryngomalacia cases were treated via supraglottoplasty with an effective rate of 83.93%. Notably, preterm birth (OR = 3.868, 95% CI 1.340 ~ 11.168), low birth weight (OR = 4.517, 95% CI 1.477 ~ 13.819) and medical comorbidities (OR = 7.219, 95% CI 2.534 ~ 20.564) were independent risk factors for poor prognosis (P < 0.05)., Conclusion: Laryngomalacia is common among infants under the age of one, and it is mostly characterized by inspiratory laryngeal stridor with various medical comorbidity. Supraglottoplasty is the first treatment choice for severe laryngomalacia cases with high success rates. However, premature delivery, low birth weight, and medical comorbidities significantly affect the efficacy of surgery., (© 2023. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

Study Objectives: Postoperative respiratory complications (PORCs) can occur following supraglottoplasty (SGP) for obstructive sleep apnea. However, there are very limited data on risk factors associated with these complications. This study aims to evaluate the occurrence of PORC in children undergoing SGP and to assess clinical factors and polysomnographic parameters predicting these complications., Methods: A retrospective study was performed in children with laryngomalacia who underwent SGP with preoperative polysomnography., Results: 400 children who underwent SGP met the criteria for entry into the analysis with a total of 416 surgeries with corresponding preoperative polysomnography. The median age (interquartile range) at the time of polysomnography was 0.4 (0.2, 1.5) years. A total of 96 (23.1%) PORCs were noted. Compared with those without complications, children with PORCs had a higher proportion of congenital heart disease ( P < .05), higher median obstructive apnea-hypopnea index (obstructive AHI; median 16.0 vs 11.4 events/h; P < .01), and lower median oxygen saturation (SpO 2 ) ( P < .001). The unadjusted odd ratio indicated an increased risk of PORCs in children with congenital heart disease (odds ratio 1.66; P < .05) and those with an obstructive AHI > 10 events/h (odds ratio 2.06; P < .01). Multiple regression analysis demonstrated that an obstructive AHI > 10 events/h was the only independent risk factor for PORCs ( P < .05)., Conclusions: In our cohort of children with laryngomalacia undergoing SGP, those with underlying congenital heart disease, higher obstructive AHI, and lower SpO 2 were more likely to develop PORCs. Only children with an obstructive AHI > 10 events/h were at significantly increased risk for PORCs following SGP. Preoperative polysomnography is useful in preoperative planning in children undergoing SGP., Citation: Kanavitoon S, Ngamprasertwong P, Gurbani N, et al. Polysomnographic parameters and clinical risk factors predicting postoperative respiratory complications in children undergoing supraglottoplasty. J Clin Sleep Med . 2024;20(1):9-16., (© 2024 American Academy of Sleep Medicine.)

Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Objectives: Laryngomalacia is the most common congenital cause of stridor; the natural history of the disease runs through to complete resolution by the age of two. Severe cases are characterized by cyanosis, hypoxia, apnea, furcular and/or subcostal retractions, aspirations, pulmonary hypertension, and failure to thrive and must undergo surgery. This study aimed to evaluate the success rates of supraglottoplasty in our hospital and evaluate the predictive factors for surgical success., Methods: Cohort study, prospectively planned. 75 patients undergoing endoscopic surgery from July 2007 to July 2016 were analyzed at the Santo Antônio Children's Hospital. The primary outcome was percentage of surgical success, defined as the absence of respiratory symptoms or presence of a mild stridor without retractions on the first post-operative month (late success). The secondary outcomes were the early surgical success (absence of respiratory symptoms or presence of a mild stridor without retractions on the first post-operative day)., Results: 39 (58.2%) were male, with an average of 4.9 months. Surgical success on the first day was 80.6% (n=54). At the end of the 1st month, surgical success was 88.6%, considering only those who completed assessment. Twenty-one (34%) presented comorbidities. Presence of comorbidities, pharyngomalacia and GERD were associated with a worse result on the 1st postoperative day, whereas, at the end of the first month, presence of comorbidities, concomitant injuries (tracheo and bronchomalacia) and pharyngomalacia were the predictive variables of surgery failure., Conclusion: Supraglottoplasty has high rates of efficacy and low morbidity. The presence of comorbidities and pharyngomalacia has shown association with a worse early and late surgical outcome. Synchronous airway lesions predict a worse surgical result at the end of the first month. GERD was associated with obstructive symptomatology only in the 1st post-operative day., Level of Evidence: Level 3 of evidence, according to the "The Oxford 2011 Levels of Evidence" from Oxford Centre for Evidence-Based Medicine., (Copyright © 2023 Associação Brasileira de Otorrinolaringologia e Cirurgia Cérvico-Facial. Published by Elsevier España S.L.U. All rights reserved.)

Objective: To characterize the prevalence and presentation of laryngomalacia and efficacy of supraglottoplasty (SGP) in a cohort of patients with Pierre Robin Sequence (PRS)., Design: Retrospective cohort study., Setting: Tertiary-care children's hospital., Patients, Participants: Consecutive patients with PRS born between January 2010 and June 2018., Main Outcome Measures: Chart review included demographics, comorbid airway obstruction including laryngomalacia, timing of surgical interventions, clinical symptoms, sleep study data, and modified barium swallow study data., 126 patients with PRS were included; 54% had an associated syndrome, 64% had an overt cleft palate, and 22% had a submucous cleft palate. 64/126 were noted to have laryngomalacia (51%). Patients with concurrent PRS and laryngomalacia were significantly more likely to have submucous cleft palate ( P  = .005) and present with aspiration with cough ( P  = .01) compared to patients with PRS without laryngomalacia. Patients with concurrent laryngomalacia and PRS showed a significant decrease in apnea-hypopnea index (AHI) and obstructive AHI (OAHI) after mandibular distraction, with a median AHI and OAHI improvement of 22.3 ( P  = .001) and 19.8 ( P  = .002), respectively. Patients who underwent only SGP did not show significant improvement in these parameters ( P  = .112 for AHI, P  = .064 for OAHI)., The prevalence of laryngomalacia in our PRS cohort was 51%. Patients with PRS and laryngomalacia are more likely to present with overt aspiration compared to patients with PRS without laryngomalacia. These data support that laryngomalacia does not appear to be a contraindication to pursuing MDO., Competing Interests: Declaration of Conflicting InterestsThe author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Objective: To analyze the judgment efficiency of a computer stress model and severity score in severity evaluation and treatment plan selection of laryngomalacia patients., Methods: Twenty-two children (12 cases in the operation group and 10 cases in the follow-up group) with moderate to severe laryngomalacia were assessed by laryngomalacia severity score (LSS) which included visual analogue scale (VAS) and clinical score. A computer stress model of the laryngeal cavity was constructed for all children, with the von Mises stress peak (VMSP) of the model used as another quantitative evaluation method. The ROC curves of two quantitative evaluation methods, the LSS and the VMSP, were analyzed respectively, according to the clinical guideline which is regarded as the gold standard for judging whether surgery is needed. The diagnostic efficiency indexes such as sensitivity, specificity, and accuracy were calculated. The area under ROC curves (AUC) of the two methods were compared by a DeLong model. Spearman correlation analysis and Kappa test were used to test the correlation and consistency of the two quantitative evaluation methods. The independent sample t test was used to compare the difference of LSS and VMSP between operation group and follow-up group., Results: The sensitivity, specificity, and accuracy of LSS in judging whether laryngomalacia was operated or not were 83.33%, 80.00% and 81.82%, respectively, and the area under ROC curve (AUC) was 0.825 (p < 0.05). The sensitivity, specificity, and accuracy of the computer stress model for laryngomalacia were 58.33%, 90.00% and 72.73%, respectively, and the AUC was 0.796 (p < 0.05). The spearman correlation coefficient between LSS and VMSP was 0.833, p < 0.001, which is statistically significant. LSS (t = 3.251, p = 0.004) and VMSP (t = 2.435, p = 0.024) of the two groups were statistically different., Conclusion: VMSP and LSS have high diagnostic efficacy in the quantitative evaluation of the severity of laryngomalacia and the selection of treatment plan. The consistency of the two quantitative evaluation methods is good, which has practical value for the evaluation of the severity of laryngomalacia and has guiding significance for surgery., (© 2023. The Author(s), under exclusive licence to Springer-Verlag GmbH Germany, part of Springer Nature.)

Background and Objectives: A large proportion of infants with vallecular cyst (VC) have coexisting laryngomalacia (LM). Feeding difficulties, regurgitation, occasional cough, and sleep-disordered breathing are the common symptoms in moderate to severe cases. The surgical management of these cases is more challenging and remains controversial. The purpose of this study is to help surgeons select the effective surgical strategies by computer-aided design (CAD) and computational fluid dynamics (CFD) simulations of the upper airway flow characteristics., Methods: The three dimensional (3D) geometric model of the upper airway was reconstructed based on two dimensional (2D) medical images of the patient with VC accompanied with LM. Virtual surgeries were carried out preoperatively to simulate three possible post-operative states in silico. The different outcomes of virtual surgical strategies were predicted based on computational evaluations of airway fluid dynamics including pressure, resistance, velocity, and wall shear stress (WSS)., Results: The CFD results of this study suggested the importance of the angle between the rim of epiglottis and arytenoid epiglottic (AE) fold. There was a small impact on the upper airway flow field while the VC was removed and the angle of epiglottis was unchanged. The partial lifting of epiglottis can further improve the flow field. With performing supraglottoplasty (SGP) and the marsupialization of VC, epiglottis was completely recovered, and the flow field was significantly improved. The clinical symptoms of this patient improved greatly after surgeries and no recurrence or growth retardation were noted during 1-year follow-up. The clinical prognosis was consistent with the prediction of the CFD results., Conclusions: The state of epiglottis needs to be carefully checked to evaluate the necessity of performing further SGP in the patients with VC accompanied with LM. CFD and CAD could be developed as a new approach to help surgeons predict the post-operative outcomes through quantification of the airflow dynamics, and make the optimal and individualized surgical approaches for patients with airway obstruction., Competing Interests: Declaration of Competing Interest The authors have no competing of interests to declare., (Copyright © 2023 Elsevier B.V. All rights reserved.)

Objective: Despite being a more accessible and less resource intensive modality than polysomnography, the utility of sleep oximetry (SO) in pediatric laryngomalacia (LM) is not well understood or validated. We aimed to retrospectively evaluate the utility of overnight home based SO in children with LM by developing and internally validating the Modified Laryngomalacia Oximetry Score (MLOS) scoring system to triage severity and guide clinical decision making., Methods: We evaluated pediatric patients with a diagnosis of LM at our tertiary referral centre. Data from initial and post-treatment SO including mean oxygen saturation (spO2) nadir and mean oxygen desaturation index (ODI) were aggregated. The MLOS ranging from I-VI (inconclusive to severe) was created by two otolaryngologists to incorporate bradycardia associated desaturation events during SO. Corresponding McGill Oximetry Score (MOS) was also determined., Results: 172 patients were included in final analysis. The average age was 9.2 ± 14.3 months. 98 (57%) of patients were identified as Thompson severity score 1, and 87 (50.6%) of patients underwent supraglottoplasty. The surgical cohort had a significantly higher MLOS and MOS scores of 4 and 2 respectively, and higher mean ODI and spO2 nadir metrics. When evaluating post-supraglottoplasty SO tracings, all parameters improved significantly, including median MLOS score from 4 to 1. Only the mean ODI improved in the non-surgical cohort. Patients with Thompson severity score 2/3 had significantly higher MLOS., Conclusion: We present a simple scoring system based on overnight SO, the MLOS, to help triage severity of pediatric LM and guide decision-making. MLOS is associated with worse clinical severity and a need for surgery, and shows significant improvement after surgery., (Copyright © 2022 Elsevier B.V. All rights reserved.)

Objective: Laryngomalacia (LM) is the most common congenital anomaly of the larynx. The cause of LM is still largely unknown, but a neurological mechanism has gained the most acceptance. There have not been any studies examining the prevalence of LM in infants with Neonatal Abstinence Syndrome (NAS). The aim of our study is to determine if infants with NAS are more likely to be diagnosed with LM., Methods: This study was a population-based inpatient registry analysis. We examined nationwide neonatal discharges in 2016 using the Kids' Inpatient Database (KID). Only patients listed as neonates were included. The International Classification of Diseases, 10th revision, Clinical Modification (ICD-10-CM) codes for neonatal withdrawal symptoms from maternal use of drugs of addiction (P96.1) and diagnoses denoting LM were used. To quantify associations between the LM and NAS groups, prevalence rates and odds ratios (ORs) were used., Results: There were 3 970 065 weighted neonatal discharges in the 2016 KID. Among patients included in our dataset, 0.809% (32 128) had NAS and 0.075% (2974) had LM. There was an increased odds ratio for neonates with NAS and LM (OR of 2.85, 95% CI = 2.24-3.63) compared to infants without NAS. Multiple logistic regression accounting for possible confounders produced an adjusted OR of 1.68 (95% CI = 1.29-2.19)., Conclusion: Our study found an association between NAS and LM. This suggests that prenatal exposure to opioids or possibly the sequelae of withdrawal symptoms may be risk factors for the development of LM.

Objective: To evaluate predictors of complications in children with congenital laryngomalacia who underwent laryngeal surgery., Methods: Multi-institutional retrospective analysis using the American College of Surgeons-NSQIP-P database (2014-2019). CPT code 31541 and ICD-10 code Q35.1 (congenital laryngomalacia) were used to select patients <18 years. Variable predictors included demographics and medical co-morbidities. Main outcomes assessed included total length of hospital stay (LOS), reintubation, reoperation and readmission., Results: 1092 children were identified, 450 (41.1%) females and 642 (58.6%) males, with a mean age of 1.1 years (95% CI 1.0-1.2). Mean LOS was 3.9 days (95% CI 3.3-4.6). Sixteen (1.5%) were reintubated, 30 (2.7%) were readmitted, and 18 (1.6%) were reoperated within 30 days. Thirty-one (2.8%) were still in hospital at 30 days. Regression analysis revealed an increase in LOS for several groups; the largest effect was seen for patients with preoperative ventilator dependence, admission within the first 28 days of life, and those who were discharged to other healthcare facilities (p < .001). Preoperative co-morbidities significantly associated with a higher frequency of reintubation included ventilator dependence (p = .003), history of prematurity (p = .016) and chronic lung disease (p = .041). Children undergoing surgery in the first 28 days of life were 10.16 times as likely to return to the OR for a related reason within 30 days than older children (p = .038)., Conclusion: Complications were rare after laryngeal surgery with a postop diagnosis of laryngomalacia. Recognizing pertinent clinical factors can help with risk assessment and management., (Copyright © 2022. Published by Elsevier Inc.)

Objective: Evidence supporting the use of acid suppression therapy (AST) for laryngomalacia (LM) is limited. The objective of this study was to determine if outpatient-initiated AST for LM was associated with symptom improvement, weight gain, and/or avoidance of surgery., Methods: A retrospective cohort was reviewed at a tertiary-care children's hospital. Patients were included if they were diagnosed with LM at ≤6 months of age, seen in an outpatient otolaryngology clinic between 2012 and 2018, and started on AST. Primary outcomes were improvement of airway and dysphagia symptoms, weight gain, and need for surgery. Severity was assessed by symptom severity., Results: Of 2693 patients reviewed, 199 met inclusion criteria. Median age of diagnosis was 4 weeks (range: 0-29 weeks). LM was classified as mild/moderate (71.4%) and severe (28.6%) based on symptom severity. Severity on flexible fiberoptic laryngoscopy (FFL) was not associated with clinical severity. Weight percentile, airway symptoms, and dysphagia symptoms improved within the cohort. In total, 26.1% underwent supraglottoplasty (SGP). In multivariate analysis, only severe LM on FFL was predictive of SGP (OR: 7.28, 95%CI: 1.91-27.67, p = .004)., Conclusion: Clinical symptom severity did not predict response to AST raising the question of utility of AST in LM. Severity of LM based on FFL, not clinical severity, was associated with decision to pursue SGP. Prospective randomized trials are needed to better understand the role of AST in LM., Level of Evidence: Level 3., (Copyright © 2022. Published by Elsevier Inc.)

Stridor is a high-pitched extrathoracic noise associated with turbulent airflow, commonly associated with respiratory distress in infants. Workup for stridor requires evaluation of the upper-respiratory airway, with severe distress requiring evaluation under anesthesia. The differential diagnosis of stridor depends on location of the obstruction, age of the patient, and acuity of the symptoms. The most common reason is laryngomalacia; most patients can be managed conservatively with resolution of symptoms by 2 years of age. In children who do not improve or have severe disease, supraglottoplasty is the treatment of choice, and the majority will have resolution of stridor postoperatively., (Copyright © 2021 Elsevier Inc. All rights reserved.)

Objective: To analyze the clinical characteristics and factors affecting surgical efficiency in children with severe laryngomalacia. Methods: Retrospectively collect medical records of children with severe laryngomalacia who underwent supraglottoplasty in Children's Hospital of Chongqing Medical University between January 1, 2015 and May 1, 2019. And analyze the clinical characteristics, the improvement of main symptoms at different time points, and the influence factors on the surgical efficiency. Results: According to the anatomical classification of severe laryngomalacia, type Ⅳ accounted for the highest proportion（66.1%）, and type Ⅰ was the lowest（3.6%）. All children had stridor and dyspnea, 82.1% cases presented feeding difficulties, and 67.9% cases presented failure to thrive. The proportion of children with medical comorbidities was 62.5%, of which congenital heart disease had the highest incidence（39.3%） The surgical efficiency of severe laryngomalacia without comorbidities was 100.0%, with one type of comorbidity was 96.0%, with multiple comorbidities was 30.0%（ P <0.01）. Stridor, dyspnea, feeding difficulties were significantly improved at one month after surgery in most cases. In the group of surgical age less than 3 months, the surgical efficiency were 61.5%, 61.5% and 69.2% at 1, 3, 6 months after surgery, respectively; the surgical efficiency of children without comorbidities were 100.0%（ P >0.05） at 1, 3 ,6 months after surgery. In the group of surgical age older than 3 months, the surgical efficiency of children were 100.0% at 1, 3, 6 months after surgery regardless with or without comorbidities. The surgical failure rate and reoperation rate in children without comorbidities was 0, but in the children with comorbidities were 22.9%（ P <0.05） and 20.0%（ P <0.05）, respectively. The surgical failure rate and reoperation rate in children with multiple comorbidities was significantly higher than children with only one comorbidity（70.0% vs. 4.0%; 60.0% vs. 4.0%, P <0.01）. The overall operation success rate was 85.7% in severe laryngomalacia children in our hospital. Conclusion: Most children with severe laryngomalacia are associated with multiple medical comorbidities, and with more complex anatomical types. Supraglottoplasty can effectively improve the symptoms in most children with severe laryngomalacia. The existence of multiple comorbidities is the main cause of surgical failure., Competing Interests: The authors of this article and the planning committee members and staff have no relevant financial relationships with commercial interests to disclose., (Copyright© by the Editorial Department of Journal of Clinical Otorhinolaryngology Head and Neck Surgery.)

The most important issue in diagnosing stridor in infants and children is determining whether it is acute or chronic. The most common cause of acute stridor is croup, a viral infection with a straightforward diagnosis when patients have typical signs and symptoms (ie, inspiratory or biphasic stridor, barking cough, hoarse voice or cry, chest wall retractions). Management of mild and severe cases includes steroids. Patients with severe croup should undergo evaluation in an urgent care center or emergency department; hospital admission may be required. When symptoms are atypical for croup, physicians should consider other causes of acute stridor, including foreign body aspiration, bacterial tracheitis, and epiglottitis. The most common cause of chronic stridor is laryngomalacia, an abnormality in the laryngeal structures that causes the collapse of supraglottic structures over the airway. Symptoms typically develop in the first 1 to 2 months of life, are positional, and do not interfere with growth and development. However, if the diagnosis is in doubt or if the laryngomalacia is severe and the patient has cyanosis, worsening stridor after feeding, or inadequate weight gain, consultation with an otolaryngology subspecialist can be helpful. Most infants outgrow symptoms as the airway enlarges, but some may need pharmacotherapy for gastroesophageal reflux disease and careful feeding until this occurs. Others may need supraglottoplasty. Other causes of chronic stridor include vocal fold paralysis, subglottic hemangiomas, and glottic webs., (Written permission from the American Academy of Family Physicians is required for reproduction of this material in whole or in part in any form or medium.)

Objective: Laryngomalacia can be an important cause of obstructive sleep apnea (OSA) in infants. Nocturnal oximetry is a cheap and safe method in comparison to polysomnography for the detection of sleep-disordered breathing. The aim of this study is to evaluate the validity of nocturnal oximetry as a diagnostic tool for OSA in infants with laryngomalacia., Methods: This retrospective study included infants with laryngomalacia and a clinical suspicion of OSA who underwent a polysomnography at the Antwerp University Hospital. The oximetry was rescored manually, blinded to the polysomnography results, according to four different scoring methods. An obstructive apnea-hypopnea index (oAHI) ≥ 2/h on polysomnography was used to define OSA., Results: This study included 53 patients with laryngomalacia (51% boys, mean age 3.72 ± 0.26 months). A diagnosis of OSA was established in 46 patients (87%) by polysomnography. Among the four different scoring methods, the scoring according to Brouillette et al., yielded the highest diagnostic accuracy with a sensitivity and specificity of 91% and 25% respectively and with a negative and positive predictive value of 25% and 91%, respectively. Correlations and the Bland-Altman plot showed a wide limit of agreement for laboratory polysomnography oAHI and nocturnal oximetry ODI., Conclusion: Our data show that overnight pulse oximetry has a high sensitivity and PPV to diagnose OSA in infants with laryngomalacia. However, the low specificity and NPV indicate that PSG is still needed to exclude OSA in cases with normal oximetry., (Copyright © 2022 Elsevier B.V. All rights reserved.)

Objectives: Laryngomalacia is the commonest cause of stridor in the paediatric population. Whilst usually self-limiting, studies have suggested that the presence of synchronous airway lesions (SALs) might be associated with more severe clinical presentation and additional morbidity. However, evidence in the literature is scarce, contributing to controversy regarding the appropriate investigation and management of children with laryngomalacia. The aim of this study was to explore the prevalence, clinical significance and risk factors of SALs in children with severe, progressive and atypical laryngomalacia symptoms., Methods: Retrospective analysis of the records of all paediatric patients diagnosed with laryngomalacia in a UK tertiary referral centre, over a 2-year period, was undertaken. Data on demographics, comorbidities, management and presence of SALs were collected., Results: The study included 130 patients with laryngomalacia. Seventy-eight patients required investigation with microlaryngobronchoscopy (MLB), based on clinical presentation. SALs were identified in 22 patients (28.2%), 6 of which (7.7%) required surgical intervention. The commonest airway lesions included tracheomalacia, bronchomalacia and subglottic stenosis. A correlation between the presence of neurodevelopmental conditions in children and SALs was noted (p value < 0.05). The presence of SALs was not associated with sex, gestational age at birth or other medical comorbidities., Conclusion: The results of this study allow professionals to counsel families about the utility of MLB in children with severe, progressive and atypical laryngomalacia symptoms and inform management pathways for clinicians not working in airway centres. We support the clinical practice that progressive, atypical or severe laryngomalacia symptoms should prompt consideration of an MLB, to identify potential synchronous airway lesions. Furthermore, we propose that a higher index of suspicion and more thorough investigation might be indicated in children with neurodevelopmental disorders., (Copyright © 2021 Elsevier B.V. All rights reserved.)

Background : Laryngomalacia is a congenital abnormality of the larynx that commonly occurs in children and rarely in adults. We report the first case of acquired laryngomalacia mainly due to postoperative seizure and central pontine myelinolysis after scheduled craniotomy. Case presentation : A 69-year-old man was admitted to the hospital for elective craniotomy for craniopharyngioma. After the surgery, he developed refractory seizure and required intubation and mechanical ventilation in the intensive-care unit (ICU). After treatment for the seizure, he was extubated. However, immediately after extubation, he developed stridor and respiratory retraction. We performed fiberoptic laryngoscopy and confirmed that the epiglottis had collapsed into the posterior wall of the pharynx during inspiration, which was suspected to be laryngomalacia. He received invasive mechanical ventilation for two days following re-extubation. After the second extubation, he developed stridor again due to acquired laryngomalacia. Six days later, his respiratory condition had worsened, and he received re-intubation and tracheostomy. After ICU discharge, central pontine myelinolysis was diagnosed by magnetic resonance imaging. Conclusions : Adult-onset laryngomalacia is a rare cause of upper airway obstruction but should be considered as a cause of postoperative extubation failure. We should not delay performing fiberoptic laryngoscopy to evaluate this pathology and provide optimal treatment. J. Med. Invest. 69 : 316-319, August, 2022.

Objective: To determine whether the use of acid suppression and thickened feeds impact laryngomalacia outcomes in infants, including supraglottoplasty risk, time to supraglottoplasty, and hospitalization risk., Study Design: We performed a retrospective cohort study to compare risk and time with supraglottoplasty and frequency and duration of hospitalizations for infants diagnosed with laryngomalacia at Boston Children's Hospital between January 1 and December 31, 2017. The primary outcomes were supraglottoplasty requirement, time to supraglottoplasty, and hospitalization risk. Multivariate analyses were performed to determine predictors of supraglottoplasty and hospitalization risk after adjusting for laryngomalacia severity and comorbidities in addition to propensity score adjustment. Kaplan-Meier curves were created to determine the impact of acid suppression use on time to supraglottoplasty., Results: In total, 236 subjects with mean age 62.6 ± 4 days were included in the analysis; 55% were treated with acid suppression. Subjects treated with acid suppression had a greater risk of supraglottoplasty (hazard ratio 3.36, 95% CI 1.36-8.29, P = .009), shorter time to supraglottoplasty (5.64 ± 0.92 vs 7.98 ± 1.92 months, P = .006), and increased respiratory hospitalization risk (relative risk 1.97, 95% CI 1.01-3.85, 0.047), even after adjustment for covariates. Subjects receiving thickening had fewer respiratory hospitalization nights and longer time to supraglottoplasty (9.3 ± 1.7 vs 4.56 ± 0.73 months, P = .004), even after adjustment., Conclusions: Acid suppression use does not reduce the frequency of supraglottoplasty and related hospitalizations compared with untreated subjects. However, patients treated with thickening have decreased hospitalization and longer time to supraglottoplasty, suggesting that thickening of feeds may be a preferred intervention over acid suppression., (Copyright © 2021. Published by Elsevier Inc.)

Laryngomalacia is the most common cause of stridor in newborns. Affected patients may present with noisy breathing, a classic high-pitched inspiratory stridor that worsens with feeding. While the exact etiology remains unclear, the condition is characterized by softening of the supraglottic structures, including the epiglottis, aryepiglottic folds, and arytenoid cartilages. The condition is most often self-limited and requires expectant management. However, in some infants, severe disease, including failure to thrive or respiratory distress, may require medical or even surgical intervention. When caring for premature neonates, special care is required to evaluate for synchronous airway lesions., (Copyright © 2021 by the American Academy of Pediatrics.)

Objective: To explore the diagnosis and therapeutic strategy of laryngomalacia associated with tongue base cyst in infants. Methods: A retrospective analysis was performed on 36 cases of tongue base cyst including 12 cases with concurrent laryngomalacia. Results: All the cysts of the tongue base were completely resected, and no recurrence occurred during the 6-months follow-up. Twelve patients with laryngomalacia were intubated in the ICU after surgery. Tube detachment occurred in one patient in the 1st day after surgery and no reintubation was performed immediately because of stable breathing and the absence of stridor. However, the stridor occurred again in the 5th day after surgery, and revision surgery was performed 1 month after surgery. One patient still had obvious stridor after extubation in the 5th day after sugery, but the shape of glottis was good by laryngoscopy, the stridor was significantly improved at one-month follow-up. One patient died in the 4th day after the operation. The efficiency of surgical treatment in pediatric patients with tongue base cyst and concurrent laryngomalacia was 83.33%. Conclusion: Laryngomalacia combined with tongue base cyst is not uncommon in clinical practice. The symptoms of tongue base cyst may mask those of the underlying laryngomalacia, since their symptoms are highly similar. Simple resection of tongue base cyst may affect the outcome of treatment. Surgeons should further investigate and identify the presence of laryngomalacia after surgical resection of the tongue base cyst and make corresponding management. For type Ⅲ laryngomalacia that cannot be determined intraoperatively, low-temperature plasma ablation of mucosa in the epiglottic valley may be a more minimally invasive option., Competing Interests: The authors of this article and the planning committee members and staff have no relevant financial relationships with commercial interests to disclose., (Copyright© by the Editorial Department of Journal of Clinical Otorhinolaryngology Head and Neck Surgery.)

Infantile haemangioma represents a congenital vascular anomaly commonly observed in the head and neck region. Such an occurrence over the postcricoid region, however, is rather unusual. Herein, the authors report a case of a synchronous postcricoid haemangioma in a 7-week-old newborn diagnosed with severe laryngomalacia. In addition to the floppy redundant arytenoid mucosa, flexible laryngoscopy revealed a lobulated bluish mass at the postcricoid. The lesion was hyperintense on T1-weighted sequence and was enhanced with contrast, supporting the diagnosis of a haemangioma. She underwent surgical excision of the haemangioma with intralesional steroid injection. Surveillance at 6-month postoperation did not show disease recurrence., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2021. No commercial re-use. See rights and permissions. Published by BMJ.)

The main objective was to analyse the use of rigid laryngotracheoscopy under general anaesthesia (GA) and endoscopic surgery in the management of inspiratory stridor in patients referred to a paediatric ENT outpatient clinic. The secondary objective was to analyse the aetiological diagnoses made and their therapeutic management. This is a prospective study including all newborns and infants, corresponding to 190 patients, presenting for the first time in consultation for inspiratory stridor from January 2015 to December 2017. A consultation form was filled out after each consultation and added to a database; a management algorithm was used to determine which patients required a rigid laryngotracheoscopy. A 17.9% (n = 34) of the patients required rigid laryngotracheoscopy, of whom 12.6% (n = 24) underwent concomitant endoscopic surgery. A 65.8% (n = 125) of the patients were diagnosed with laryngomalacia, 21.1% (n = 40) with isolated posterior excess of mucosa, 9.5% (n = 18) with another diagnosis and 3.7% (n = 7) with a normal examination. The presence of comorbidity was associated (p < 0.001) with the use of rigid laryngotracheoscopy and endoscopic surgery.Conclusion: Rigid laryngotracheoscopy under GA was required in one in five to six patients. Conservative management with strict follow-up may be appropriate in a large number of patients, especially those with laryngomalacia. What is Known: • Previous research has established that laryngomalacia is the main aetiology of stridor. • Comorbidities are linked with a poor tolerance of stridor. What is new: • About one in five to six patients seen in consultation for stridor will require a trip to the operative room (and one in eight will require endoscopic surgery). • Laryngomalacia and isolated posterior excess of mucosa account for 85-90% of the patients seen in consultation for stridor.

Introduction: Children with Down syndrome (DS) who have Laryngomalacia represent a specific management challenge due to their inherent multiple levels of airway obstruction and hypotonia. Therefore, the purpose of this study is to determine the outcome of supraglottoplasty (SGP) in children with DS., Data Source: A systematic review was performed following the PRISMA guideline. Data were collected from online medical databases- CINAHL, Cochrane Library, Google Scholar, Ovid MEDLINE, Ovid MEDLINE in process, PubMed, and Scopus., Review Methods: all publications that address the outcome of supraglottoplasty in children who are less than 18 years old with Down syndrome prior to December 2018 were collected. The data were collected on different phases: Screening review using search words and controlled vocabularies during the period of November-December 2018, followed by a detailed review of screened articles based on inclusion and exclusion criteria, then a full review that included screening the references of selected articles., Results: Nine articles were included in the qualitative analysis. These articles included 231 subjects, out of which 32 children with DS met the inclusion criteria. Twenty patients (62.5%) were considered successfully treated and 12/32 failed including 1/32 (3.1%) mortality in the postoperative period. The duration of follow-up was 12-102 days. All of the patients whose treatment failed had significant comorbidities., Conclusion: Although the success of SPG in children with DS is less than in otherwise healthy children, it remains an option especially in patients without comorbidities. Preoperative counseling of the families and thorough assessment are necessary to determine candidacy for SPG in children with DS., (Copyright © 2021 Elsevier B.V. All rights reserved.)

Sublingual dermoid cysts and oral heterotopic gastrointestinal cysts are well-documented causes of sublingual swellings in infants. However, the simultaneous occurrence of both cysts is an extremely rare phenomenon, with only two previous cases reported in literature. We present a case of 1-month-old neonate with a cystic ventral tongue mass that on histologic examination showed a dermoid cyst with co-existing heterotopic gastric-type epithelium in the wall. Additional histochemical stains revealed focal areas of colonic metaplasia within the heterotopic gastric epithelium. CDX2 immunohistochemical staining confirmed derivation from colonic epithelium.

Objectives: Laryngomalacia (LM) is commonly diagnosed in infants and children with upper aerodigestive symptoms. In the literature, the focus has been on the respiratory impairment, with fewer studies addressing swallowing dysfunction (SwD). The objective of this study is to systematically review the literature for evidence on the prevalence of SwD in children diagnosed with LM., Methods: A search was conducted on the following databases: OVID Medline, Ovid EMBASE, EBSCO CINAHL, PROSPERO, and Cochrane Library. We included all the studies that reported on children with LM and documented objective swallowing assessment using fiberoptic endoscopic evaluation of swallowing (FEES) or videofluoroscopic swallowing study (VFSS). Two authors independently screened all the studies, assessed the level of evidence in the included studies, and extracted data. Risk of bias assessment and pooled data analysis were performed., Results: The search yielded 512 abstracts. Four studies met the selection criteria representing 425 children. Three studies were retrospective uncontrolled case series and one was a prospective cohort study. In all studies but one, an instrumental assessment of swallowing was selectively performed in patients with clinical indicators of SwD. The pooled estimate (range) of prevalence of SwD was 49% (13.9-90.6%)., Conclusion: The literature suggests a high prevalence of SwD in children with LM, however the level of evidence is low and generalizability is poor. The wide range of prevalence figures suggests a significant variability in the threshold and indications to assess swallowing in children with LM., (Copyright © 2020 Elsevier B.V. All rights reserved.)

Introduction: Obstructive sleep apnea (OSA) in young children is caused by upper airway obstruction and is associated with changes in cognitive development, temperament and behavior. Adenotonsillectomy (AT) is often utilized as first line therapy for pediatric OSA. Children with laryngomalacia (LM) have a high chance of residual OSA after AT. There is paucity of literature regarding surgically naïve young children with OSA and laryngomalacia. Our study aimed to compare demographics, comorbidities and outcomes associated with OSA in surgically naïve young children with and without laryngomalacia., Methods: Retrospective chart review of surgically naïve young children (<2-year-old) with polysomnogram (PSG) diagnosed OSA. All young children underwent pre-operative PSG followed by drug-induced sleep endoscopy (DISE) directed intervention. Variables documented included demographics, comorbidities, history of adenotonsillectomy, DISE directed surgical interventions and pre and post PSG findings. Laryngomalacia was defined as presence of obstruction (Chan Parikh score ≥ 2) at the supraglottic level on DISE evaluation. Demographics and prevalence of comorbidities of those with and without LM were compared using t-test (continuous) and Chi Square (categorical). P value is significant for <0.05., Results: 79 surgically naïve young children with PSG diagnosed OSA performed between 2015 and 2019 were included in the study. Children with LM were younger in age (11 months) and had a higher OSA-18 score (35) compared to children without LM (17 months) (OSA-18 score: 5). No significant difference was noted in the pre-DISE PSG parameters in the 2 groups. 56.1% of children with OSA and laryngomalacia did not need AT. Both children with and without laryngomalacia showed improvement in AHI on post-DISE PSG. Higher baseline AHI was associated with greater improvement in AHI., Conclusion: Surgically naïve young children with OSA and LM present earlier than those without LM and report a significantly worse quality of life Young children with a higher baseline AHI were found to have greater improvement in severity of OSA post-DISE and surgical intervention. Majority of children with OSA and laryngomalacia did not need adenotonsillectomy after a preprocedural DISE assessment yet showed similar improvement in AHI. More studies are needed to determine which patients with OSA and laryngomalacia will need multilevel interventions., (Copyright © 2020 Elsevier B.V. All rights reserved.)

Introduction: Laryngomalacia is one of the most common causes of stridor in young children. It can be a serious concern to both parents and caregivers. The main objective of this study is to find the prevalence of laryngomalacia among young children presenting with stridor in a tertiary care hospital in central Nepal., Methods: A descriptive cross-sectional study was carried out form 1st December 2017 to 1st May 2020 in children less than two years of age in a tertiary care hospital. Ethical approval was taken from the Institutional Review Committee of the hospital (reference number: 2020/23). Convenient sampling was done. Detailed demography, clinical examination, and video laryngoscopy findings were evaluated to find the prevalence of laryngomalacia among all children with stridor. Data were analyzed by using Statistical Package for the Social Sciences version 20. Point estimate at 95% confidence interval was calculated along with frequency and proportion for binary data., Results: Out of 430 participants who presented with stridor, the laryngomalacia was found in 234 (66%) (58.7-74.07) cases at a 95% confidence interval. The male: female ratio was 1.7:1. Most children, 192 (67.6%), presented with a milder form of laryngomalacia. The most common type was a mixed type of laryngomalacia in 159 (56%). Sleep-disordered breathing was seen in 113 (39.79%) of children diagnosed with laryngomalacia., Conclusions: Our study concluded that laryngomalacia was the most common cause of stridor in children less than two years of age. However, in most cases, the problem is not serious and a regular follow-up with weight monitoring is warranted.

Objectives: Children with laryngomalacia may present with obstructive sleep apnea (OSA). The role of polysomnography (PSG) in treatment decision making for laryngomalacia is not well defined. We aimed to investigate the prevalence of OSA in children with laryngomalacia and the role of PSG in treatment decision., Methods: Retrospective medical record review of children with laryngomalacia, confirmed by direct laryngoscopy, during a period of 3 years. Demographic data, presenting symptoms, severity classification, comorbidities and pre- and postoperative PSG data were retrieved and analyzed. Data are expressed as a median (25th - 75th percentile)., Results: Forty-six patients were with diagnosed laryngomalacia between March 2016 and April 2019. A complete data set was available for 44 patients, 24 males and 20 females. The median age at the time of PSG was 12 weeks (6.3-29.8). Thirty-four children (77.4%) were diagnosed with concomitant OSA. A diagnosis of OSA changed the severity classification and treatment decision in 24 cases (54.5%). Twenty-three patients underwent supraglottoplasty, five patients were treated with continuous positive airway pressure (CPAP) and nine patients had both treatments. Seven patients received conservative treatment. The obstructive apnea/hypopnea index decreased from 8.9 events/hour (4.4-12.1) to 2.4 events/hour (1.5-4.4) after supraglottoplasty (p = 0.009)., Conclusions: A diagnosis of OSA was established in 77.4% of patients with larygomalacia The presence of OSA may increase the severity of symptoms in laryngomalacia, leading to a transition from watchful-waiting to active intervention with CPAP therapy or supraglottoplasty. Supraglottoplasty is a safe and effective surgical procedure for laryngomalacia. When performed in the setting of laryngomalacia with concomitant OSA, it also significantly improves OSA symptomatology., (Copyright © 2020. Published by Elsevier B.V.)

Objective: Endoscopic supraglottoplasty is the procedure of choice in treatment of laryngomalacia with CO2 laser and cold steel being the most common instruments used with excellent results. However, bleeding, supraglottic stenosis and aspiration may occur leading to serious complications. Using coblation in management of laryngomalacia was found to be beneficial. Therefore, we conducted this study to evaluate the outcome of supraglottoplasty done by coagulation using coblation of the lateral surface of aryepiglottic folds as an alternative technique to aryepiglottic fold release in management of type 2 laryngomalacia., Methods: Retrospective study was conducted at Mansoura University Hospitals; Egypt from November 2017 to March 2018 included patients diagnosed with severe type 2 laryngomalacia. Supraglottoplasty was done by using coblator applied to the lateral surface of aryepiglottic folds allowing for lateral scarring thus widening the airway, preventing supraglottic collapse and avoiding re-stenosis. Outcome measures included assessment of presence or absence of the following symptoms preoperatively and postoperatively: inspiratory stridor, failure to thrive, choking and cyanosis. Preoperative and postoperative lowest oxygen saturation levels, weight-by-age percentile, and need for tracheostomy were assessed., Results: Nine patients were included in this study with a mean age of 3.78 ± 1.20 months (range 2-6 months). Overall success rate was 89%. The most significantly improved symptom was stridor (p-value 0.008). Significant improvement in lowest oxygen saturation levels (from 89.11 ± 3.06% pre-operatively to 96.44 ± 3.50% post-operatively) and weight gain (from 4288.9 ± 643.1 gm. preoperatively to 5505.55 ± 1017.4 gm. 1 month postoperatively) was achieved. No detected re-stenosis on follow up and none of our cases needed tracheostomy., Conclusion: Supraglottoplasty by coagulation of the lateral surface of aryepiglottic folds using coblation is an effective and safe technique and can improve airway symptoms and weight gain in patients with type 2 laryngomalacia., Competing Interests: Declaration of Competing Interest None., (Copyright © 2019. Published by Elsevier B.V.)

Objective: Laryngomalacia is the most common cause of infant stridor, and obstructive sleep apnea (OSA) is sometimes found concurrently in patients with laryngomalacia. OSA has been shown to improve after surgical treatment of laryngomalacia, but the majority of laryngomalacia patients have spontaneous resolution of symptoms. It is unknown whether their comorbid OSA also resolves. This study seeks to define the incidence of OSA in laryngomalacia and assess for resolution of OSA with polysomnography data., Methods: Retrospective cohort study at a tertiary care academic medical center. All pediatric patients with diagnoses of laryngomalacia or stridor were reviewed, and patients with laryngomalacia confirmed by Otolaryngologist exam were included. All patients with laryngomalacia were recommended to undergo polysomnography., Results: A total of 108 patients had laryngomalacia confirmed by an Otolaryngologist. Of those patients, 56 completed a polysomnogram, and 44 (79%) were diagnosed with OSA. Among the OSA patients, 34 had no surgery, 5 underwent supraglottoplasty, and 5 underwent adenoidectomy or adenotonsillectomy. Follow-up polysomnograms were performed for 9 non-surgical patients, 4 supraglottoplasty patients, and 4 adenoidectomy or adenotonsillectomy patients. Mean change in AHI was -2.81 without surgery, -8.18 after supraglottoplasty, and -2.94 after adenoidectomy or adenotonsillectomy., Conclusion: OSA is often present in patients who have laryngomalacia, and the proportion in this population was higher than previous reports. The only significant predictor for obstructive sleep apnea was race, specifically Black/African American. Among patients with follow-up polysomnograms, the largest OSA improvement was in supraglottoplasty patients, but all patients improved., Competing Interests: Declaration of competing interest No conflicts of interest or competings interests exist for the authors in this study., (Copyright © 2020 Elsevier B.V. All rights reserved.)

Objectives/hypothesis: Laryngomalacia is the most common cause of stridor in infants. The exact pathophysiology is still not well understood. Our objective was to investigate whether laryngomalacia is an inflammatory disease, focusing on the possible role of vitamin D., Study Design: Case-control study., Methods: Sixty Egyptian infants and 60 mothers were included in this study. They were divided into four equal groups (n = 30 for each): infants with laryngomalacia (LM-infants), control infants (C-infants), mothers of the infants with laryngomalacia (LM-mothers), and mothers of the control infants (C-mothers). Laryngoscopy was performed and serum 25-hydroxyvitamin D (25[OH]-vitamin-D) and interleukin 6 (IL-6) were estimated., Results: Significant increase of serum IL-6 associated with a significant decrease in serum 25(OH)-vitamin D was observed in the LM-infants compared to the C-infants (P < .001 for both). LM-mothers had significantly lower 25(OH)-vitamin D status compared to C-mothers (P < .001)., Conclusions: Deficiency of 25(OH)-vitamin D in LM-infants may result in dysregulation of the immune responses with elevation of a proinflammatory cytokine (IL-6). Laryngomalacia could be an inflammatory disease due to 25(OH)-vitamin D deficiency as evidenced by the high level of IL-6. This finding may open the door to the appropriate prevention, diagnosis, and treatment, especially for moderate to severe laryngomalacia., Level of Evidence: 3b Laryngoscope, 130:448-453, 2020., (© 2019 The American Laryngological, Rhinological and Otological Society, Inc.)

Objectives: Laryngomalacia is an established cause of stridor and sleep-disordered breathing in children. However, the relationship between laryngomalacia and dysphagia has not been well characterized. The objectives of this study were to (1) describe the patient characteristics, symptoms, and prevalence of dysphagia in children with laryngomalacia and (2) examine the effectiveness of supraglottoplasty in improving feeding., Methods: This was a retrospective study of patients with laryngomalacia who underwent a modified barium swallow study (MBSS) at a tertiary academic pediatric medical center between March 1, 2014, and March 1, 2018. Patients were excluded if they did not undergo a MBSS. Comorbidities, airway and feedings symptoms, MBSS results, and surgical history were recorded from each patient's electronic medical record., Results: Forty-four children met inclusion/exclusion criteria. The median age at presentation was 96 days. There was a male predominance (66%). About one-third had a genetic or neuromuscular comorbidity. Most children had stridor (93%) and feeding difficulty (86%), while 50% had parent-reported poor weight gain. Fifty-seven percent of patients had evidence of penetration or aspiration on MBSS. All patients with a positive MBSS had dysphagia symptoms. Fifty-seven percent of patients underwent supraglottoplasty. Postoperatively, 92% reported improvement in dysphagia symptoms., Conclusion: Dysphagia is prevalent among a subset of children with laryngomalacia. Symptomatic children may benefit from a swallow evaluation to help determine the need for further intervention. Children who undergo supraglottoplasty for laryngomalacia have improved dysphagia at follow-up, though the amount of improvement directly attributable to surgery is unclear and warrants further investigation.

Objective: To perform a systematic review with meta-analysis of data to determine the rates of repeat surgery and supraglottic stenosis in unilateral versus bilateral supraglottoplasty for laryngomalacia., Data Sources: PubMed/Medline, Cochrane Central, Scopus, Google Scholar, Web of Science, and Embase., Review Methods: Databases were searched through January 30, 2018. Studies with unilateral or bilateral supraglottoplasty techniques for laryngomalacia were included. The need for repeat (revision or completion) surgery and rates of supraglottic stenosis were primary outcomes. Data were substratified and a meta-analysis performed., Results: A total of 251 articles were reviewed, and 20 articles met inclusion criteria (1186 patients: 663 bilateral, 523 unilateral). Regarding the need to return to surgery, the rate of revision for bilateral surgery was 4.1%, compared to the revision and combined revision/completion rates for unilateral surgery which respectively were 1.1% (odds ratio [OR] 0.27; 95% CI 0.11-0.67; P = .002) and 18.0% (OR 5.16; 95% CI 3.31-8.06; P < .0001). The unilateral versus bilateral supraglottic stenosis rates were 0% versus 1.2% ( P = .011)., Conclusion: Unilateral supraglottoplasty has a significantly higher rate of repeat surgery, mainly attributed to contralateral surgery, when compared with bilateral supraglottoplasty. There is a small but statistically significant risk of supraglottic stenosis in bilateral procedures. The benefit of a unilateral procedure should be weighed against the cost of subjecting patients to a 4-fold increased risk of repeat surgery.

Objective: Laryngomalacia has been reported to contribute to the severity of obstructive sleep apnea (OSA) in children. It is unclear if surgical treatment of laryngomalacia improves polysomnography (PSG) outcomes in these patients. The objective of this study is to report the impact of supraglottoplasty on PSG parameters in children with laryngomalacia-related OSA., Study Design: Retrospective case series., Setting: Tertiary care medical center., Subjects and Methods: Historical cohort study of consecutive children with laryngomalacia who underwent supraglottoplasty and who had undergone overnight PSG before and after surgery., Results: Forty-one patients were included in the final analysis: 22 (53.6%) were male, and 19 (46.3%) were female. The mean ± SEM age of patients at preoperative PSG was 1.3 ± 0.89 years (range, 0.003-2.9). In entire cohort, the mean obstructive apnea-hypopnea index score was reduced from 26.6 events/h before supraglottoplasty to 7.3 events/h after surgery ( P = .003). Respiratory disturbance index was reduced from 27.3 events/h before supraglottoplasty to 7.8 events/h after surgery ( P = .003). The percentage of REM sleep decreased from 30.1% ± 2.4 to 24.8% ± 1.3 ( P = .04). Sleep efficiency was improved ( P = .05)., Conclusion: Overall, supraglottoplasty significantly improved several PSG outcomes in children with laryngomalacia. However, mild to moderate OSA was still present postoperatively in most children. This suggested a multifactorial cause for OSA in this population.