Transient swallowing dysfunction in typically developing children following supraglottoplasty for laryngomalacia

ObjectiveTo evaluate early postoperative swallowing function in otherwise typically-developing children following supraglottoplasty.MethodsRetrospective chart review case series.ResultsOf 37 children identified as having undergone supraglottoplasty for severe laryngomalacia at our institution between January 2007 and October 2011, 24 were identified as otherwise typically developing with no indications/signs of swallowing problems pre-operatively and eligible for inclusion in this study. Twenty-two children underwent bilateral supraglottoplasty and 2 children underwent unilateral supraglottoplasty using the CO₂ laser or the laryngeal skimmer microdebrider in combination with cold steel technique, based on the discretion of four Pediatric Otolaryngologists. Seventeen children were seen post-operatively for a clinical swallow evaluation. Six children were found to have swallowing dysfunction. Four of the six children showed variable symptoms, signs, or findings concerning for aspiration with oral feeding. Three of six underwent video fluoroscopic swallow study (VFSS). All 6 children responded to dietary modifications, positioning alterations, and anti-reflux medications. All showed improvement by their 4-week post-operative follow-up visit. The median age of the 24 patients at the time of surgery was 3 months, with a range from 2 weeks to 4 years. The median age of the patients found to have transient post-operative swallowing dysfunction was 1.5 months, with a range of 2 weeks to 4 months. There was no association between post-operative swallowing dysfunction and the surgical technique employed.ConclusionsSupraglottoplasty in otherwise typically developing children carries a risk of transient swallowing dysfunction with a low risk of persistent dysfunction. Most patients can be assessed post-operatively via clinical swallow evaluation without requiring an instrumental swallow study.     

Feasibility of Surgical Treatment for Laryngomalacia Using the Spontaneous Respiration Technique

Objectives.In this study, we review our institutional experience with pediatric laryngomalacia (LM) and report our experiences of patients undergoing supraglottoplasty using the spontaneous respiration using intravenous anesthesia and high-flow nasal oxygen (STRIVE Hi) technique.Methods.The medical records of 29 children with LM who visited Seoul National University Hospital between January 2017 and March 2019 were retrospectively reviewed. Surgical management was performed using the STRIVE Hi technique. Intraoperative findings and postoperative surgical outcomes, including complications and changes in symptoms and weight, were analyzed.Results.Of the total study population of 29 subjects, 20 (68.9%) were female. The patients were divided according to the Onley classification as follows: type I (n=13, 44.8%), II (n=10, 34.5%), and III (n=6, 20.7%). Twenty-five patients (86.2%) had comorbidities. Seventeen patients (58.6%) underwent microlaryngobronchoscopy under STRIVE Hi anesthesia. Four patients with several desaturation events required rescue oxygenation by intermittent intubation and mask bagging during the STRIVE Hi technique. However, the procedure was completed in all patients without any severe adverse effects. Overall, 15 children (51.7%) underwent supraglottoplasty, of whom 14 (93.3%) showed symptom improvement, and their postoperative weight percentile significantly increased (P=0.026). One patient required tracheostomy immediately after supraglottoplasty due to associated neurological disease.Conclusion.The STRIVE Hi technique is feasible for supraglottoplasty in LM patients, while type III LM patients with micrognathia or glossoptosis may have a higher risk of requiring rescue oxygenation during the STRIVE Hi technique.     

Supraglottoplasty outcomes in children with Down syndrome

ObjectiveChildren with Down syndrome have a higher incidence of upper airway obstruction and laryngomalacia. We sought to determine outcomes of supraglottoplasty in this patient population.MethodsA retrospective chart review was performed from January 2000 through January 2014. Children (n = 18) at our institution with the diagnosis of Down syndrome who underwent supraglottoplasty were included. We reviewed patient characteristics, preoperative findings, and surgical outcomes (stridor, feeding problems, respiratory distress, weight, sleep apnea, and tracheostomy or feeding tube dependence).ResultsThe average age at surgery was 7.7 months. Operative indications included feeding difficulties (n = 9), noisy breathing or respiratory distress (or both) (n = 16), and sleep-related symptoms (n = 7). Most patients (89%) were extubated successfully on postoperative day 1. There were 2 major complications (CPAP requirement and aspiration pneumonia) and no perioperative deaths. Fifty percent had improved weight (mean = 18 percentile points). Feedback was available from 88% of parents with 100% reporting improvement in respiratory symptoms and 93% reporting improved feeding. Eight patients (44%) subsequently required either adenoidectomy or adenotonsillectomy. Two patients later underwent tracheostomy, 2 subsequently needed a gastrostomy tube and 2 required revision supraglottoplasty.ConclusionsThe majority of children with Down syndrome and laryngomalacia benefit from supraglottoplasty, with outcomes of improved breathing, feeding, and sleeping.However, approximately half may require additional airway procedures. This procedure is well tolerated and associated with a low risk of complications especially given their high rate of comorbidities.     

Late-onset laryngomalacia: a cause of pediatric obstructive sleep apnea

Objective

To describe the presentation, diagnosis, and treatment of late-onset laryngomalacia in children with obstructive sleep apnea syndrome (OSAS).

Design

Retrospective study.

Setting

Tertiary care children's hospital.

Patients

Seventy-seven children were identified who had OSAS diagnosed by polysomnography and underwent airway endoscopy to evaluate for laryngomalacia between July 2006 and December 2008. Children with significant neurologic disease or craniofacial malformations were excluded. Seven children under 3 years of age had laryngomalacia and OSAS (Group A), 19 children 3-18 years of age had laryngomalacia and OSAS (Group B), and 51 children 3-18 years of age had OSAS but not laryngomalacia (Group C).

Main outcome measures

Comparison of pre-operative findings, intra-operative findings, interventions, and outcomes between the 3 groups.

Results

Group A was consistent with previous reports of congenital laryngomalacia with respect to presentation, diagnosis, and treatment. Groups B and C had similar pre-operative findings, including a high incidence of adenotonsillar hypertrophy, and the only significant difference was the intra-operative finding of laryngomalacia in Group B. Treatments were individualized to include supraglottoplasty, adenoidectomy, tonsillectomy, adenotonsillectomy, or a combination of the above. Of the 52 patients who returned in follow-up, 44 noted improvement, but this was rarely confirmed by polysomnogram.

Conclusions

Late-onset laryngomalacia may act alone or in concert with additional dynamic or fixed lesions to cause pediatric OSAS. Although there is no specific pre-operative indicator to diagnose late-onset laryngomalacia, it can be readily identified intra-operatively and effectively treated with supraglottoplasty, with or without concurrent adenotonsillectomy.     

Otolaryngological presentations of Cornelia de Lange syndrome

Aim

Children with Cornelia de Lange syndrome frequently present to otolaryngology services with hearing problems. Airway problems have not previously been reported. We wish to describe our experience of the overall management in a series of children with Cornelia de Lange syndrome.

Methods

Retrospective case note review of children diagnosed with Cornelia de Lange syndrome presenting to our department between 2005 and 2014.

Results

Six patients were seen. Airway problems consisted of laryngeal overspill with severe gastroesophageal dysmotility and reflux despite structurally normal airway (1 case), laryngomalacia requiring supraglottoplasty (2 cases), reflux laryngitis with secondary laryngomalacia and coincidental tracheal diverticulum (1 case) choanal atresia requiring stents (1 case) and obstructive sleep apnoea (1 case). Supraglottoplasty produced a dramatic improvement in feeding and breathing in both children who underwent the procedure. Two children had palatal anomalies and one underwent cochlear implantation for a profound sensorineural hearing loss.

Conclusion

Children with Cornelia de Lange syndrome have multifaceted ENT problems. Airway pathology has not previously been described in Cornelia de Lange syndrome but has been common in our experience. We wish to highlight that laryngomalacia in Cornelia de Lange syndrome responds well to supraglottoplasty.     

Risk factors for failure of supraglottoplasty

Objectives

To assess for identifiable risk factors for failed surgical intervention in children with laryngomalacia.

Methods

Retrospective case note review between September 2007 and March 2012. Case notes were reviewed for demographic data, symptoms, co-morbidities, operative technique, postoperative recovery, complications, length of hospital stay including intensive care unit (ICU) care, and resolution of symptoms.

Results

148 children underwent supraglottoplasty. Case notes were available for 115 (78%) patients. 35% (41/115) of cases were females and 65% (74/115) were male. A bimodal age distribution was observed with peaks at 3 months and 3.5 years. Those over one year of age were more likely to have complications (p = 0.035). There was no significant difference in outcomes for age (p > 0.05). In patients less than one year, reflux symptoms were significantly associated with a higher likelihood of failure of the operation (p = 0.013). Patients under one year with pre-operatively identified comorbid conditions were less likely to have an improvement in breathing (p = 0.002). Cold steel was used in 55% (63/115) of cases, laser only in 17% (20/115) cases and a combination of the two techniques in 28%, (32/115). There was no association between the surgical technique used and complications (p = 0.558). There was no association between improvement in symptoms and surgical technique used (p = 0.560). There was a significant association between delayed post-operative neurological diagnosis and failure of the operation (p < 0.001). 21 (18%) patients required a second procedure.

Conclusions

Pre-operative predictors of failure were patients with reflux symptoms (p = 0.013). Patients that required a second procedure were 37 times more likely to have a delayed diagnosis of an underlying neurological condition. Failure of symptoms to improve after supraglottoplasty should alert the clinician to the possibility of an underlying neurological disorder.     

Characteristics of patients undergoing supraglottoplasty for laryngomalacia

Objective

To examine the characteristics of patients undergoing supraglottoplasty for the treatment of laryngomalacia and to better understand the features of laryngomalacia that may predispose patients to the need for supraglottoplasty.

Methods

Review of patients who underwent supraglottoplasty for laryngomalacia at our academic tertiary care children's hospital between 2005 and 2012 examining demographic information, medical comorbidities, symptoms, indications for surgery, operative findings and procedure, site of laryngeal obstruction, operative techniques, and surgical success rates.

Results

Seventeen patients with laryngomalacia underwent nineteen procedures. The most common indications for supraglottoplasty were persistent stridor beyond 18 months of age (64.7%), difficulty feeding (47%), and failure to thrive (29.4%). The most common comorbidities were gastroesophageal reflux (88.2%) and cardiopulmonary disease (35.3%). Operative findings included shortened aryepiglottic folds in sixteen patients (94.1%), retropositioned epiglottis in sixteen (94.1%), and prolapsed arytenoid mucosa in five (29.4%). Fifteen patients (88.2%) underwent division of the aryepiglottic folds and four underwent ablation of arytenoid mucosa (23.5%). Of the seventeen patients who had followed up at the time of study conclusion, sixteen (94.1%) had symptom improvement and twelve (70.6%) had complete resolution of their symptoms.

Conclusions

Supraglottoplasty is an effective treatment for laryngomalacia. Outcomes in our patients are similar to those reported in prior literature. The findings of shortened aryepiglottic folds and a retropositioned epiglottis appear to be disproportionately represented in our cohort of patients undergoing supraglottoplasty.