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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).)

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.)

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.)

Objectives: To evaluate the role infant pulmonary function tests (Tidal Breathing Flow Volume Loops, TBFVL) in children with airway anomalies and to correlate the TBFVL so obtained with bronchoscopy findings., Methods: In this prospective cohort study, we enrolled children aged 0-2 years with airway anomalies and performed TBFVL and bronchoscopy. The primary outcome measure was graphic pattern of TBFVL in laryngomalacia. Secondary outcome measures were types of TBFVL results in various airway anomalies and controls., Results: Out of 53 children enrolled, 28 (52.3%) had laryngomalacia. Pattern 3 (fluttering of inspiratory limb) was commonest TBFVL pattern in laryngomalacia. Among TBFVL parameters, the ratio of inspiratory time to expiratory time (Ti/Te) and tPTEF/tE was significantly high in children with isolated laryngomalacia compared to controls. At six months of follow-up, TBFVL pattern 1 (normal) became the commonest pattern., Conclusion: A particular type of airway anomaly may have a characteristic graphic pattern in TBFVL and TBFVL pattern may indicate improvement in airway anomalies in follow-up.

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: 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.)

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.)

Congenital laryngomalacia is the most common disease causing laryngeal stridor in infants. The pathogenesis has not yet been clearly concluded. It may be related to abnormal development of laryngeal cartilage anatomical structure, neuromuscular dysfunction, gastroesophageal and laryngeal reflux disease, etc. The typical manifestations of the disease are inspiratory laryngeal stridor and feeding difficulties, which can be divided into mild, moderate and severe according to the severity of symptoms. The diagnosis is mainly based on clinical symptoms, signs and endoscopy, among which endoscopy is an important diagnostic basis. The treatment of laryngomalacia depends on the severity of symptoms. Mild and some moderate congenital laryngomalacia children can be relieved by conservative treatment, and severe and some moderate congenital laryngomalacia children should be treated by surgery. Supraglottic plasty is the main surgical method, which can effectively improve the symptoms of laryngeal stridor, dyspnea, feeding difficulties and growth retardation in most children, and the surgical effect is good., 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.)

Background: Severe laryngomalacia, characterized by apnea, hypoxia, and feeding difficulties, is an uncommon diagnosis that often requires surgical intervention with supraglottoplasty. Children who require surgery at a young age and those with additional comorbidities pose a special challenge and may require further surgical interventions. Posterior displacement of the epiglottis has been noted in some infants with congenital stridor and is commonly treated with epiglottopexy. The goal of our study is to review the outcomes of epiglottopexy combined with supraglottoplasty in our cohort of infants younger than 6 months old with severe laryngomalacia., Methods: A retrospective chart review of infants younger than 6 months old who underwent epiglottopexy combined with supraglottoplasty for severe laryngomalacia from January 2018 to July 2021 at a tertiary care children's hospital., Results: 13 patients (age 1.3 week-5.2 months) underwent supraglottoplasty and epiglottopexy for severe laryngomalacia and epiglottis retroflection. The patients were admitted to the intensive care unit and remained intubated for at least one night. All patients demonstrated subjective and objective improvement in upper airway respiratory signs and symptoms. Ten patients demonstrated aspiration immediately postoperatively, despite 4 of them having no concern for aspiration at preoperative evaluation. On follow-up, 1 patient required revision supraglottoplasty and epiglottopexy for persistent laryngomalacia, and 2 patients required tracheostomy tube placement due to cardiopulmonary comorbidities., Conclusion: Infants younger than 6 months old with medical comorbidities undergoing epiglottopexy with supraglottoplasty may demonstrate significant improvement in respiratory symptoms. Worsening dysphagia may complicate the postoperative period, particularly among children with medical comorbidities.

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/aim: The pathology of laryngomalacia is still not clear. The aim of this study was to investigate the relationship between vitamin D levels and laryngomalacia, and to evaluate vitamin D levels according to the classification of laryngomalacia., Materials and Methods: This retrospective study was conducted in the Kahramanmara ş S ütçü İ mam University Medicine Faculty's Otorhinolaryngology Clinic between June 2014 and January 2021. Laryngomalacia was classified. Laboratory tests for all patients included calcium (Ca), phosphorus (P), parathormone (PTH), blood urea nitrogen (BUN), creatinine (Cre), alanine transaminase (ALT), and 25-hydroxy vitamin D (25-OH-D)., Results: Evaluations were performed for 64 infants with laryngomalacia, including 41 male and 23 female infants with a mean age of 4.6 ± 3.0 months, and a control group of 64 healthy infants with a mean age of 4.5 ± 2.8 months. A statistically significant difference was determined between the laryngomalacia group and the control group with respect to 25-OH-D and PTH levels (p < 0.001). When data were examined according to laryngomalacia types, a statistically significant difference was determined between the groups for 25-OH-D, Ca, P, PTH, and ALT values. The 25-OH-D level was statistically significantly lower in the severe laryngomalacia group than in the mild and control groups (p < 0.001). A statistically significant difference was determined between the moderate and severe laryngomalacia groups and the control group regarding PTH levels (p < 0.001)., Conclusion: Vitamin D deficiency may have a role in the etiology of laryngomalacia, and this view is supported by the finding that there was a decrease in vitamin D levels associated with laryngomalacia classification. In addition, the reduction in PTH levels in infants with laryngomalacia may be explained by the change in Ca metabolism. It would be appropriate for further studies to investigate the response to vitamin D replacement therapy in patients with moderate and severe laryngomalacia., Competing Interests: Conflict of interest: The authors declared no conflict of interest., (© TÜBİTAK.)

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.)

Objectives: To compare patient specific and surgical efficiency outcomes in children undergoing supraglottoplasty (SGP) with flexible fiber CO2 laser in Ultrapulse mode versus cold steel at a tertiary care center., Methods: A single surgeon retrospective cohort study of pediatric patients with laryngomalacia undergoing SGP without concomitant or prior airway surgery. We reviewed perioperative complications, total hospital stay including intensive care admission, symptom resolution and the need for revision surgery. Surgical efficiency was measured as the mean total operative time and laryngeal suspension time., Results: Among 63 patients, 32 cold steel SGPs were compared with 31 flexible fiber CO2 Ultrapulse laser SGPs. There was no statistical difference in the demographic profile. There was no statistical difference in need for intraoperative resuscitation (1 [3.0%] vs 0 [0%] cases) or perioperative intubation (4 [12.1%] vs 2 [6.3%] cases), mean total days in ICU (1.09 [0.51] vs 1.06 [0.44] [p = 0.38]) or days hospitalized (1.73 [1.67] vs 1.62 [2.27] [p = 0.42]), symptom resolution, need for revision surgery (2 [6.3%] vs 0 [0%] [p = 0.49] patients) or mean operative time (29.0 [14.19] vs. 32.2 [22.71] [p = 0.95] minutes). There was a statistically significant decreased time in laryngeal suspension in the laser group versus cold steel group (5.9 [1.80] vs 7.9 [2.19] [p = 0.006] minutes)., Conclusion: For pediatric SGPs, optimal flexible fiber CO2 laser usage can improve surgical efficiency in comparison to cold steel, with no differences in postoperative outcomes or complications. Both the flexible fiber CO2 laser and cold steel SGPs are safe and effective methods for treatment of laryngomalacia., (Copyright © 2022 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: 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.)

Introduction: As a recently established division, we sought to reflect on the development of our paediatric airway surgery service, and prospectively examine the diagnoses that underwent microlaryngobronchoscopy (MLB) to help quantify the evolving population demographics of paediatric airway disorders., Material and Methods: This was a prospective longitudinal study conducted of all paediatric MLBs performed by a single surgeon in a tertiary paediatric ENT centre between 2012 and 2019., Results: A total of 1040 MLBs were performed in 498 patients at the paediatric ENT centre of the Royal London Hospital. Median age at first procedure was 19 months. Median follow-up was 48 months. Primary diagnoses were laryngomalacia (21%), subglottic stenosis (SGS - 18%), laryngeal cleft (13%), and normal anatomy (28.3%). Repeat procedures were needed in 39.1% patients, who underwent a median of 2 repeat procedures. SGS (57.7%) constituted majority of the repeat category, followed by laryngeal cleft (12.36%), laryngomalacia (10.15%), unilateral/bilateral vocal cord palsy(4.24%) and laryngeal papilloma(4.24%). Laryngeal papilloma constituted largest number of procedures per patient (Median = 4, IQR = 5.75), followed by subglottic web and SGS. Mean length of stay(LOS) was 0.67 ± 0.96 days(d), with laryngeal cleft cases recording longest mean LOS. There was a steady increase in proportion of day-surgeries across study period [6.9% (2012) vs 59%(2019)]., Conclusion: SGS constitutes the major bulk of paediatric airway surgery, reflective of increasing number of premature births and prolonged intubation among neonates. Day-case MLB is a safe and feasible option in selected patients. This long-term data provides useful information to accurately prognosticate patients regarding potential number of repeat procedures for each diagnosis., (Copyright © 2022 Elsevier B.V. All rights reserved.)

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.)

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.

Background: Flexible fiber-optic laryngo-tracheo-bronchoscopy has become widely performed in infants and neonates since the introduction of thin flexible fiberscopes. Laryngomalacia is the most common airway disease in infants causing stridor. Pharyngomalacia, termed pharyngeal occlusion during inspiration, was the second most common airway disease found in our hospital in patients that underwent laryngo-tracheo-bronchoscopy, but the incidence, patient characteristics, and natural course have not been reported in large numbers in Japan., Methods: A retrospective review was performed of medical records on patients admitted to our neonatal intensive care unit during the neonatal period diagnosed with pharyngomalacia between April 2009 and November 2018. Patient characteristics, concurrent airway diseases, comorbidities, and treatment were reviewed., Results: Forty-eight patients were diagnosed with pharyngomalacia. The median gestational age was 37.1 weeks, and the median birthweight was 2,552 g. Patients were diagnosed at a median age of 29 days, and cure was achieved at a median age of 4 months. Twenty-eight patients had concurrent airway diseases, laryngomalacia being the most common. Continuous positive airway pressure or high flow nasal cannula was used in 34 patients., Conclusions: In patients with pharyngomalacia, half were born preterm, and more than half had concurrent airway diseases. The onset and diagnosis were made within the first month of life in more than half of the patients, and resolution was seen mostly within the first 6 months of life. Whenever a patient is suspected of having an airway disease, the pharyngeal space should be carefully observed to diagnose pharyngomalacia., (© 2021 Japan Pediatric Society.)

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.)

Background: Infantile tremor syndrome (ITS) is a rare clinical entity, predominantly characterized by coarse tremors, regression of motor and/or cognitive, and language milestones. Although the exact pathogenesis yet remains to be unknown, various micronutrient deficiencies like vitamin B12, zinc, magnesium, and vitamin C have been shown to be associated with ITS. Vitamin B12 deficiency is the most accepted etiology of this entity. Here we are describing a seemingly novel association of laryngomalacia with infantile tremor syndrome., Methods: Clinical details, laboratory investigations, demographic and socioeconomic parameters of all children<2 years of age, diagnosed with ITS between August 2019 and November 2020 was obtained by a retrospective chart review. The study population was divided into two subgroups based on the presence/absence of laryngomalacia and different variables were compared between the two subgroups., Results: During the study period, 22 ITS and 13 pre-ITS cases were identified, out of which 5 ITS cases had laryngomalacia, the prevalence is higher as compared to estimated prevalence in reported literature (p < 0.0001). All these 5 cases had associated gastro-esophageal reflux and swallowing dysfunction and all of them were managed conservatively, apart from standard care treatment for ITS. On follow-up, all 5 of them showed improvement in symptoms of laryngomalacia, apart from clinical and hematological improvement. Differences between various clinical and biochemical parameters in the subgroup with and without laryngomalacia were not significantly different. Seventeen out of twenty-two children with ITS and 10/13 children with pre-ITS had macrocytic anemia and the rest had dimorphic anemia on peripheral smear. There was moderate vitamin B12 deficiency in 13/22 and 8/13 children with ITS and pre-ITS respectively, and the rest had a severe vitamin B12 deficiency., Conclusion: Clinicians need to screen cases with ITS for probable symptoms of laryngomalacia and accordingly advise management measures. Although our study showed a seemingly apparent association between ITS and laryngomalacia, large prospective controlled studies with long-term follow-up are necessary to prove this association., Competing Interests: Declaration of competing interest All the authors declare that they have no conflict of interest., (Copyright © 2021 European Society for Clinical Nutrition and Metabolism. Published by Elsevier Ltd. All rights reserved.)

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.)

Competing Interests: Competing interests: None declared.

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.)

Objective: To evaluate which factors may affect anxiety and child's health perception of parents with children that have laryngomalacia., Study Design: Survey Study., Setting: "Coping with Laryngomalacia" - largest online laryngomalacia support group for parents with children who have laryngomalacia., Subjects: and Methods: A survey was sent to support group member that gathered information regarding the patient's diagnosis and associated course of treatment and evaluated for parental anxiety and perceived health of their child. Analysis was performed to evaluate which variables are associated with increased parental anxiety and worsened perceived child's health., Results: Data was gathered on 434 patients. All caregivers reported some level of anxiety due to the diagnosis and 64% felt completely anxious. Parents who saw an increased number of physicians prior to a pediatric ENT rated their child with poorer health (p < .05). Those that felt their concerns were brushed off initially reported increased anxiety (p < .05). If the child was admitted to the hospital, ICU, required intubation, had feeding troubles, required a feeding tube, or underwent pH monitoring, the patient was rated to have poorer health per the parents (p < .05)., Conclusion: Laryngomalacia causes a significant burden on patients and their families due to the eating problems, feeding problems, hospital stay, and other various conditions associated with the disease. Caregivers should take a more modern and compassionate approach to management and diagnosis., (Copyright © 2020 Elsevier B.V. All rights reserved.)

Objectives: Endoscopic supraglottoplasty is the mainstay surgical procedure in treatment of laryngomalacia. Various instruments have been used with coblation recently gaining widespread attention. Regarding the technique used, cutting the aryepiglottic folds is associated with rapid improvement but carries risk of restenosis while outer scar contracture method has delayed but established result. Therefore, this study was conducted to introduce a modified coblation supraglottoplasty technique gathering both benefits and evaluate its results and effectiveness., Methods: Retrospective study included patients diagnosed with type II laryngomalacia was conducted. Supraglottoplasty was done by "one and half coblation supraglottoplasty" technique which involves cutting of one aryepiglottic fold while the other one is laterally coagulated. Patients' basic and clinical data were assessed. Outcome measures included assessment of inspiratory stridor, failure to thrive, choking, cyanosis, lowest oxygen saturation levels and weight gain., Results: Seventeen patients were included in this study with a mean age of 3.71 ± 1.1 months. Significant statistical improvement was reached regarding stridor, failure to thrive and cyanosis. Also, minimal oxygen saturation and weight gain were significantly improved. None of the cases needed tracheostomy and no major postoperative complications occurred., Conclusion: We conclude that "one and half coblation supraglottoplasty" is an effective and safe treatment for type II laryngomalacia with satisfactory outcomes., Level of Evidence: IV., (Copyright © 2020 Elsevier B.V. All rights reserved.)

Competing Interests: The authors of this article and the planning committee members and staff have no relevant financial relationships with commercial interests to disclose.

Objectives: Laryngomalacia is the dynamic collapse of supraglottic structures during inspiration, leading to a variable degree of airway obstruction. Clinical symptoms appear in the first months of life and are usually mild and resolve by the age of 12-18 months. In severe cases, surgical intervention may be considered. The goal of the study was to review the clinical outcome of pediatric patients who underwent supraglottoplasty for laryngomalacia., Material and Methods: Clinical and demographic data were retrieved from medical records of children diagnosed with laryngomalacia by laryngo-bronchoscopy between 2013 and 2019. Indications, outcome and long-term follow-up were collected from children undergoing surgery., Results: During the study period, 115 children were diagnosed with laryngomalacia. The median age at diagnosis was 3 months. Synchronous airway lesions were diagnosed in 20% of patients. Ten (8.7%) children underwent surgical treatment because of significant respiratory symptoms and/or failure to thrive. Three of them had comorbidities. All otherwise healthy children had significant respiratory and nutritional improvement after surgery while those with comorbidities had less successful outcomes., Conclusion: We conclude that in severe cases of laryngomalacia, supraglottoplasty has an important role to play in management. In children with comorbidities, the surgical results may be less successful. Therefore, we recommend that the decision to operate should be individualized, ensuring full disclosure to the family regarding the probable benefit along with the limitations of surgery., (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.)

We report a case of CHARGE syndrome with atypical phenotype and a novel mutation in the CHD7 gene. Laryngomalacia and swallowing difficulties are prominent features in this case. These are commonly found in patients with CHARGE syndrome and are well described in previous studies. However, with the traditional diagnostic criteria, diagnosis is difficult without the presence of coloboma or choanal atresia. Early diagnosis is possible with the aid of clinical genetics. The current diagnostic criteria would need to be broadened with the inclusion of pathogenic CHD7 variant status as a major criterion. Further research on the function of CHD7 gene may also give us more insight on the pathogenic mechanism of various clinical features of CHARGE syndrome., Competing Interests: Competing interests: None declared., (© BMJ Publishing Group Limited 2020. No commercial re-use. See rights and permissions. Published by BMJ.)

Objective: Adult-onset laryngomalacia is a rare clinical entity that has been infrequently reported. This study aims to evaluate the clinical presentation, diagnosis, and management of adult-onset laryngomalacia through literature review and report of a case., Methods: PubMed and Google Scholar databases were queried for articles published from 1960 to 2019 including only patients aged 18 years and older. Included keywords were: 'laryngomalacia', 'adult laryngomalacia', 'acquired laryngomalacia', 'idiopathic laryngomalacia', 'laryngeal obstruction', 'floppy epiglottis', 'floppy epiglottis', and 'epiglottis prolapse'. Data extracted from literature included clinical presentation, diagnostic workup, surgical management, and follow-up care., Sources: PubMed and Google Scholar., Results: A total of 21 articles reported 41 cases of adult-onset laryngomalacia. Within these cases, 5 etiologies were identified: neurologic (n = 14), exercise-induced (n = 9), post-operative (n = 7), idiopathic (n = 7), and age-related (n = 4) laryngomalacia. Anterior prolapse of arytenoids and aryepiglottic folds was the most common laryngoscopic finding (n = 21), followed by posterior epiglottic prolapse (n = 20). Management included supraglottoplasty (n = 14), epiglottidectomy (n = 8) or epiglottopexy (n = 2). Neurologic etiology required tracheotomy more often than the other etiologies (n = 5, 36% vs. 15%). Three patients were managed expectantly without surgical intervention and reported symptom resolution., Conclusion: Adult laryngomalacia is a rare diagnosis comprising a spectrum of disease. This diagnosis may be overlooked, but association with neurologic injury or trauma should encourage consideration. In comparison to pediatric laryngomalacia, patients often require surgical intervention. Surgical decision is based on the direction of supraglottic collapse, where supraglottoplasty and partial epiglottidectomy are effective interventions., Level of Evidence: N/A., Competing Interests: Declaration of competing interest None., (Copyright © 2020. Published by Elsevier Inc.)

Objective: Vallecular cyst coexisting with laryngomalacia (VC-LM) can cause significant pharyngolaryngeal obstruction. Traditionally, it is diagnosed with flexible endoscopy (FE) and treated by rigid endoscopy. This study evaluates the effectiveness of solely using FE with novel noninvasive ventilation (NIV) of sustained pharyngeal inflation (SPI) support for both diagnosis and treatment in such infants., Methods: A retrospective review of consecutive infants who were diagnosed and treated for VC-LM in the 12-year period, 2007 to 2018, was conducted. Clinical variables, techniques, and outcomes were analyzed and reported., Results: Eighteen infants (10 males) were included. The mean age was 3.0 ± 0.6 months and the mean body weight was 4.6 ± 1.3 kg. Before FE, 14 infants were supported with bi-nasal prongs NIV (BN-NIV) and four infants with tracheal intubation. During diagnostic and therapeutic FE, all infants supported with a nasopharyngeal NIV (NP-NIV) only. All diagnoses were made in the first FE inspection of 3.5 ± 1.2 minutes. Thirteen lesions were immediately treated with FE laser therapy in 18.1 ± 1.7 minutes in the same FE course. Total FE time was 24.6 ± 2.8 minutes. Three infants needed revision laser therapy 4 days later. There was no desaturation (<90%), bradycardia (<100/min), or pneumothorax. After FE therapy, all infants were supported with BN-NIV only with significantly (<0.01) lower pressure and completely weaned off before being discharged 8.4 ± 1.5 days later. All infants, followed up for a 6-month period, showed many clinical improvements., Conclusions: FE, with this NP-NIV and SPI supports, could offer accurate diagnosis and successful laser therapy of the VC-LM with procedural sedation in the same session in infants., (© 2020 Wiley Periodicals, Inc.)

This study was performed to investigate the feasibility of using a three-dimensional (3D) finite element model for laryngomalacia severity assessment. We analyzed laryngeal computed tomography images of seven children with laryngomalacia using Mimics software. The gray threshold of different tissues was distinguishable, and a 3D visualization model and finite element model were constructed. The laryngeal structure parameters were defined. The peak von Mises stress (PVMS) value was obtained through laryngeal mechanical analysis. The PVMS values of the laryngeal soft tissue and cartilage scaffolds were independently correlated with disease severity. After stress loading the model, the relationship between laryngomalacia severity and the PVMS value was apparent. However, the PVMS value of laryngeal soft tissue was not correlated with laryngomalacia severity. This study established the efficacy of a finite element model to illustrate the morphological features of the laryngeal cavity in infants with laryngomalacia. However, further study is required before widespread application of 3D finite element modeling of laryngomalacia. PVMS values of the laryngeal cartilage scaffold might be useful for assessment of laryngomalacia severity. These findings support the notion that structural abnormalities of the laryngeal cartilage may manifest as quantifiable changes in stress variants of the supraglottic larynx.

Objectives: Examine outcomes of patients 1 month of age or less who received supraglottoplasty. Demonstrate the feasibility and safety of supraglottoplasty in neonates <1 month of age., Methods: Charts were reviewed from 2015-2017. Patients with previously identified laryngomalacia requiring surgical intervention and age 1 month or less were identified. We collected data on age, gender, surgical technique, other airway lesions, time to extubation and discharge, and comorbidities., Results: Six patients met the inclusion criteria of which four were male and two were female. Average age at time of surgery was 19.5 days. All patients had diagnosis of laryngomalacia made on flexible or direct laryngoscopy with increased work of breathing, reports of cyanotic episodes, or oxygen desaturations. All had feeding difficulties, and three had signs and history consistent with gastroesophageal reflux. Two patients underwent supraglottoplasty by CO 2 laser treatment alone, one patient received cold steel treatment alone, and the remaining three patients received cold steel with supplemental microdebrider treatment. Out of six patients, five were extubated within 5 days of their procedure. The average time to extubation was 1.8 days. The average time to discharge was 24.25 days following surgery. Two patients required revisions., Conclusions: Supraglottoplasty in children younger than 1 month of age is rare. Despite the rare occurrence, it appears to be feasible early in life in treating laryngomalacia. Neonates with respiratory failure, apneas, and cyanosis, or difficulty feeding due to laryngomalacia should be evaluated and treated with supraglottoplasty.

Objective: To correlate the clinical severity of laryngomalacia (LM) with endoscopic findings, swallowing evaluations and polysomnography in a cohort of patients. Method: We conducted a retrospective analysis between 2017-2018 on a cohort of patients diagnosed with upper airway obstruction (UAO), stridor, noisy breathing or laryngomalacia. This study took place at the Pediatric Pulmonology Department, Riley Children's Hospital, Indianapolis, United States of America., Results:   There were 157 patients with laryngomalacia included in the study. Patients with severe LM were significantly younger than those with mild LM (p=0.0214) and moderate LM (p=0.0220). Subjects with type I of LM were significantly older than type III (p=0.0051). When associations were tested between polysomnogram (PSG) variables and clinical severity, there were significant associations with age at PSG. The overall apnea-hypopnea index (AHI) in mild (p=0.0103) and moderate (p=0.0242) were significantly lower than the severe group. The rapid eye movement (REM) AHI was significantly lower in moderate cases than severe (p=0.0134). The end-tidal carbon dioxide (EtCO2) peak was significantly lower in mild cases than severe (p=0.0141). The total sleep time (TST) peripheral capillary oxygen saturation (SpO2) 90% occurs in both mild (p=0.0197) and moderate (p=0.0498) were significantly lower than the severe group., Conclusions: The severity of the clinical manifestations of LM did not correlate with the different endoscopic types in our study. The presence of cyanosis was associated with type III LM. Rapid eye movement AHI and EtCO2 in polysomnogram were remained significantly associated with clinical severity.

Objective: Supraglottoplasty is the mainstay of surgical treatment for laryngomalacia, and is commonly performed via two methods: cold steel or carbon dioxide (CO 2 ) laser. The degree of post-operative monitoring following supraglottoplasty varies, both within and between institutions. The aim of this study was to compare the post-operative monitoring and interventions required by patients undergoing cold-steel versus CO 2 laser supraglottoplasty., Design: Retrospective cohort of pediatric patients (age < 18 years) undergoing supraglottoplasty at a tertiary care pediatric hospital. The primary exposure was the surgical instrument(s) used during supraglottoplasty. The primary outcome was prolonged intensive care unit (ICU)-stay (defined as >24 h)., Results: 155 cases were eligible for inclusion. Fifty-eight (37.4%) patients had a comorbid condition. Common indications for surgery included feeding difficulty (56.1%), severe respiratory distress (33.5%), and obstructive sleep apnea (25.2%). CO 2 laser was employed in 49 cases and cold-steel in 106 cases. Prolonged ICU-stay (>24 h) was observed in 14 CO 2 laser cases (28.6%) and 11 cold-steel cases (10.4%) (adjusted OR 3.42; 95% CI 1.43, 8.33). CO 2 laser cases were more likely to require post-operative intubation, non-invasive positive pressure ventilation, and nebulized racemic epinephrine. Concomitant neurological condition was associated with an increased risk of prolonged ICU-stay, while extent of surgery and age were not., Conclusions: CO 2 laser supraglottoplasty is associated with an increased risk of prolonged ICU-stay and need for ICU-level airway intervention, compared to the cold-steel technique. While this association should not be misconstrued as a causal relationship, the current study demonstrates that specific surgical factors may influence the patient monitoring requirements following supraglottoplasty, particularly the choice of instrument and the extent of surgery., Competing Interests: Declaration of competing interest None., (Copyright © 2019 Elsevier B.V. All rights reserved.)

Objectives: Laryngomalacia is a common cause of stridor in infants and is associated with laryngopharyngeal reflux (LPR). Although pepsin in operative supraglottic lavage specimens is associated with severe laryngomalacia, detection of pepsin in oral secretions has not been demonstrated in an outpatient setting., Methods: Children <2 years old with laryngomalacia diagnosed by flexible laryngoscopy and children without stridor were selected. Oral secretion samples were obtained in clinic from all subjects. Pepsin, IL-1β, and IL-8 enzyme-linked immunosorbent assays were performed to determine presence of LPR., Results: Sixteen laryngomalacia and sixteen controls were enrolled. Pepsin was detected more frequently in oral secretions of patients with laryngomalacia (13/16) than in controls (2/16; P  < .001). Four patients with laryngomalacia developed symptoms requiring supraglottoplasty. Presence and level of salivary pepsin was not significantly associated with need for surgical management, nor were the levels or presence of IL-1β or IL-8 significantly associated with presence or level of pepsin, diagnosis of laryngomalacia, or need for operative management., Conclusion: Pepsin in saliva appears to be associated with laryngomalacia, suggesting a role for salivary pepsin as a noninvasive marker of LPR in patients with laryngomalacia. Future studies will determine the utility of this test in laryngomalacia.

We herein report the successful management of a condition mimicking acquired laryngomalacia using conservative methods in an elderly man with a progressive neurological disorder. The patient developed stridor and was transferred to the intensive-care unit. Flexible laryngoscopy revealed a collapsed epiglottis during inspiration, as seen in acquired laryngomalacia, with mucinous material firmly adhered to the epiglottis. The stridor resolved after the removal of this material. Pathology revealed keratinized material, suggesting a collection of sputum or epithelial tissue. Thus, flexible laryngoscopy can differentiate the cause of airway obstruction and avoid unnecessary endotracheal intubation in patients with neurological disorders.

The authors present the case of a 5-year-old girl referred to our institution due to several episodes of nocturnal stridor with ocular retroversion and parental notion of apnea. She has been previously submitted to adenotonsillectomy. Due to symptoms worsening she was referred to our hospital. Here, a nasal fiberoptic endoscopy evaluation was conducted and a diagnosis of laryngomalacia was done. The was submitted to CO2 laser ariepiglotoplasty with symptom improvement after surgery. During a follow-up appointment, parents reported self-limited clonic facial movements at sleep onset. The electroencephalogram (EEG) was compatible with benign childhood epileptiform discharges., (Copyright © 2019 Elsevier B.V. All rights reserved.)