ABSTRACT
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.
Subject(s)
Anesthesia, General , Bronchoscopy , Respiratory Sounds , Humans , Respiratory Sounds/etiology , Male , Infant , Anesthesia, General/methods , Laryngomalacia/complications , Laryngomalacia/diagnosis , Laryngomalacia/surgery , Tracheomalacia/complications , Tracheomalacia/diagnosis , Propofol , Ketamine/therapeutic useABSTRACT
OBJECTIVES: Patients with severe laryngomalacia (LM) and high-risk comorbidities require surgical interventions such as supraglottoplasty. However, evidence supporting epiglottopexy for these patients is scarce. This study aims to report the surgical outcomes of external double needle puncture using a single thread for epiglottopexy in severe LM patients with high-risk comorbidities. METHODS: This retrospective study was conducted at a single tertiary hospital. We enrolled 32 patients under 12 months with severe LM who underwent external double needle puncture epiglottopexy. We compared clinical factors between the successful and failed groups and identified risk factors for the failure of epiglottopexy. RESULTS: Of the airway surgical outcomes, 22 (68.7 %) patients were in the successful group. In the failed group (n = 10, 32.3 %), patients received tracheostomies due to uncontrolled saliva (n = 3), the need for mechanical ventilation (n = 3), and uncontrolled LM (n = 4). There was a statistically significant difference in body weight at the time of surgery and the proportion of associated comorbidities between the two groups. Gestational age was the only factor significantly associated with successful surgical outcomes in both univariate and multivariate logistic regression analyses (odds ratio = 2.263; 95 % confidence interval, 1.042-4.918; P = 0.039). CONCLUSION: External double needle puncture epiglottopexy is an effective surgical method for patients with LM who present with a retroflexed floppy epiglottis and high-risk comorbidities. Low gestational age is a major risk factor for surgical failure. Consideration of factors such as the need for mechanical ventilation and uncontrolled saliva should be prioritized before and after surgery to enhance surgical success.
Subject(s)
Epiglottis , Laryngomalacia , Punctures , Humans , Retrospective Studies , Male , Laryngomalacia/surgery , Female , Infant , Epiglottis/surgery , Treatment Outcome , Infant, Newborn , Comorbidity , Needles , Risk Factors , Severity of Illness IndexABSTRACT
OBJECTIVE: Laryngomalacia is the most common pediatric laryngeal anomaly. The pathophysiology of laryngomalacia is not well defined; the leading hypothesis suggests weak laryngeal tone and neuromuscular discoordination. Only a few studies explored the histopathology of the laryngeal submucosal nerves, with reported nerve hypertrophy. Our study aims to describe the histopathology of submucosal nerves in specimens obtained from children with severe laryngomalacia compared to pediatric cadaveric controls. STUDY DESIGN: Prospective study. SETTINGS: Tertiary care children's hospital. METHODS: Histologic and immunohistochemical sections of supraglottic tissue from 26 children with severe laryngomalacia and six pediatric autopsies were digitally scanned and assessed with image analysis software (QuPath), resulting in the identification and measurement of 4561 peripheral nerves and over 100,000 foci of neurofilaments. RESULTS: Chronic inflammation was noted in all patients. Eosinophils were rare. The mean nerve area and perimeter were significantly smaller for patients with laryngomalacia compared to the control group (1594.0 ± 593.2 µm^2 vs. 2612.1 ± 2824.0 µm^2, p < 0.0001, and 158.8 ± 30.3 µm vs. 217.6 ± 165.0 µm, p < 0.0001). Nerve-per-area unit was significantly greater for patients with laryngomalacia compared to controls (1.39E-05 vs. 6.19 E-06, p = 0.009). The mean area and the number of neurofilaments per total nerve area were similar. Immunohistochemistry for calretinin, a marker for intestinal ganglion cells in Hirschsprung disease, was absent from all specimens. CONCLUSIONS: This series includes a comparison of all identifiable nerve fibers obtained from children with severe laryngomalacia and shows that the mucosal nerves are smaller on average than controls. These findings fail to provide support for significant morphologic peripheral nerve pathology in laryngomalacia.
Subject(s)
Laryngomalacia , Humans , Laryngomalacia/pathology , Male , Female , Prospective Studies , Infant , Child, Preschool , Immunohistochemistry , Infant, Newborn , Autopsy , Case-Control Studies , Laryngeal Nerves/pathology , Laryngeal Mucosa/pathology , Child , Severity of Illness IndexABSTRACT
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.
Subject(s)
Laryngomalacia , Humans , Male , Female , Laryngomalacia/surgery , Laryngomalacia/complications , Infant , Treatment Outcome , Cohort Studies , Child, Preschool , Neurodevelopmental Disorders/etiology , Neurodevelopmental Disorders/epidemiology , Child Development , Conservative Treatment , Severity of Illness Index , Infant, NewbornABSTRACT
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.
Subject(s)
Laryngomalacia , Vocal Cord Paralysis , Infant , Infant, Newborn , Humans , Vocal Cords , Laryngomalacia/complications , Laryngomalacia/diagnosis , Laryngomalacia/surgery , Respiratory Sounds/etiology , Vocal Cord Paralysis/complications , Vocal Cord Paralysis/diagnosis , Deglutition , Retrospective StudiesABSTRACT
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, CI95 [1.6; 2.8], p ≤ 10-4). The weight gain rate was predicted (r2 = 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.
Subject(s)
Laryngomalacia , Polysomnography , Sleep Apnea, Obstructive , Humans , Laryngomalacia/complications , Laryngomalacia/diagnosis , Retrospective Studies , Polysomnography/methods , Male , Infant , Female , Sleep Apnea, Obstructive/therapy , Sleep Apnea, Obstructive/diagnosis , Sleep Apnea, Obstructive/physiopathology , Severity of Illness Index , Weight GainABSTRACT
BACKGROUND: The Koolen-de Vries syndrome (KdVS) is a relatively new rare disease caused by micro-deletion of 17q21.31 which was first reported by Koolen in 2006. Typical phenotypes for KdVS include hypotonia, developmental delay, moderate intellectual disability, and characteristic facial dysmorphism. Up to now, there was only one case report about anesthesia management of patient diagnosed KdVS. It was a 2-year-old girl who experienced an MRI exam under anesthesia. CASE PRESENTATION: We described a 21-month-old boy who planned to undergo an orchidopexy under general anesthesia diagnosed with KdVS. He had an intellectual disability, characteristic facial dysmorphism, tracheo/laryngomalacia, patent foramen ovale, and cryptorchidism related to KdVS. Due to the complex condition especially the presence of tracheo/laryngomalacia, we took some special measures, including reducing the amount of long-acting opioid, keeping the spontaneous breath, performing a caudal block, and applying the laryngeal mask. But the laryngeal mask was changed to an endotracheal tube because it failed to provide adequate ventilation. The boy experienced mild laryngeal spasm and hypoxia after extubation, but lateral position and etomidate eased his breathing problem and re-intubation was avoided. It is indicated that anesthesia management for patients with orphan disease is a real challenge for all anesthesia providers. CONCLUSIONS: The Koolen-de Vries syndrome is a relatively new orphan disease involving multiple systems. Keeping spontaneous breath, evaluating airway potency to anesthetics, applying endotracheal tube, and post-extubation lateral or prone position may be helpful for airway management for patient with hypotonia and tracheo/laryngomalacia. KdVS patient needs prolonged post-anesthesia monitoring and/or medication for airway complications.
Subject(s)
Abnormalities, Multiple , Chromosome Deletion , Intellectual Disability , Laryngomalacia , Humans , Infant , Male , Anesthesia, General , Chromosomes, Human, Pair 17 , Muscle Hypotonia , Rare DiseasesABSTRACT
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.
Subject(s)
Polysomnography , Sleep Apnea, Obstructive , Humans , Sleep Apnea, Obstructive/therapy , Sleep Apnea, Obstructive/diagnosis , Male , Female , Retrospective Studies , Infant , Laryngoscopy , Severity of Illness Index , Child, Preschool , Treatment Outcome , Hypertrophy , Laryngomalacia/complications , Laryngomalacia/therapy , Endoscopy , Infant, NewbornABSTRACT
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.
Subject(s)
Deglutition Disorders , Larynx , Humans , Deglutition Disorders/etiology , Deglutition Disorders/diagnosis , Larynx/abnormalities , Larynx/surgery , Child , Infant , Laryngomalacia/complications , Laryngomalacia/surgery , Laryngomalacia/diagnosis , Gastroesophageal Reflux/complications , Gastroesophageal Reflux/diagnosis , Laryngeal Diseases/surgery , Laryngeal Diseases/complications , Vocal Cord Paralysis/etiology , Deglutition/physiology , Congenital AbnormalitiesABSTRACT
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.
Subject(s)
Bronchoscopy , Respiratory Function Tests , Humans , Bronchoscopy/methods , Infant , Prospective Studies , Male , Female , Respiratory Function Tests/methods , Infant, Newborn , Child, Preschool , Laryngomalacia/diagnosis , Laryngomalacia/physiopathology , Respiratory System Abnormalities/diagnosis , Respiratory System Abnormalities/physiopathology , Tidal Volume/physiologyABSTRACT
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.
Subject(s)
Sleep Apnea, Obstructive , Humans , Infant , Infant, Newborn , Continuous Positive Airway Pressure , Laryngomalacia/diagnosis , Laryngomalacia/complications , Laryngomalacia/therapy , Polysomnography , Sleep Apnea, Obstructive/therapy , Sleep Apnea, Obstructive/diagnosisABSTRACT
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.
Subject(s)
Laryngomalacia , Humans , Laryngomalacia/surgery , Laryngomalacia/complications , Infant , Child, Preschool , Child , Treatment Outcome , Infant, Newborn , Female , MaleABSTRACT
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.
Subject(s)
Laryngomalacia , Humans , Laryngomalacia/diagnosis , Laryngomalacia/complications , Laryngomalacia/surgery , Adult , Laryngoscopy , Respiratory Sounds/etiologyABSTRACT
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.
Subject(s)
Laryngomalacia , Larynx , Premature Birth , Infant , Child , Humans , Infant, Newborn , Male , Female , Laryngomalacia/complications , Laryngomalacia/diagnosis , Laryngomalacia/surgery , Retrospective Studies , Respiratory Sounds/etiology , Treatment Outcome , Glottis/surgeryABSTRACT
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.
Subject(s)
Deglutition Disorders , Gastroesophageal Reflux , Laryngomalacia , Infant , Infant, Newborn , Humans , Deglutition Disorders/epidemiology , Deglutition Disorders/etiology , Infant, Premature , Laryngomalacia/complications , Cross-Sectional Studies , Intensive Care Units, Neonatal , Sucking Behavior , Risk Factors , Ataxia/complicationsABSTRACT
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.
Subject(s)
Laryngomalacia , Sleep Apnea, Obstructive , Infant , Humans , Infant, Newborn , Laryngomalacia/complications , Laryngomalacia/diagnosis , Laryngomalacia/surgery , Polysomnography/methods , Sleep Apnea, Obstructive/diagnosis , Sleep Apnea, Obstructive/surgery , Sleep , Endoscopy/methods , Retrospective StudiesABSTRACT
Laryngomalacia is the most frequent congenital laryngeal anomaly. Surgery is possible by means of cold knife, carbon dioxide (CO2 ) laser, microdebrider and coblation, traditionally under microscopic view. We here describe the use of the 3D-4K exoscope assisted CO2 laser supraglottoplasty in tubeless general anesthesia in spontaneous breathing in a 5 month-old patient with severe laryngomalacia. Laryngoscope, 134:1359-1362, 2024.
Subject(s)
Laryngomalacia , Larynx , Lasers, Gas , Humans , Child , Infant , Laryngomalacia/surgery , Carbon Dioxide , Retrospective Studies , Lasers, Gas/therapeutic use , Treatment Outcome , Glottis/surgeryABSTRACT
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 (SpO2) (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 SpO2 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.
Subject(s)
Heart Defects, Congenital , Laryngomalacia , Sleep Apnea, Obstructive , Child , Humans , Infant , Laryngomalacia/complications , Laryngomalacia/surgery , Retrospective Studies , Sleep Apnea, Obstructive/complications , Sleep Apnea, Obstructive/surgery , Postoperative Complications/etiology , Risk FactorsABSTRACT
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.
Subject(s)
Laryngomalacia , Child , Humans , Infant , Laryngomalacia/surgery , Laryngomalacia/complications , Retrospective Studies , Hospitalization , Critical Care , Intensive Care Units , Treatment OutcomeABSTRACT
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.