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Irace AL, Dombrowski ND, Kawai K, et al. Evaluation of Aspiration in Infants With Laryngomalacia and Recurrent Respiratory and Feeding Difficulties. JAMA Otolaryngol Head Neck Surg. 2019;145(2):146–151. doi:10.1001/jamaoto.2018.3642
What is the prevalence of aspiration in pediatric patients with laryngomalacia who present with recurrent respiratory issues and/or feeding difficulty in 1 year?
In this study of the medical records of 142 infants with laryngomalacia who presented with recurrent respiratory issues and/or feeding difficulties, aspiration was identified in 42.3% of patients, and almost all of these patients aspirated silently.
Swallowing discoordination and aspiration were common in patients with laryngomalacia and recurrent respiratory and/or feeding difficulty; these children should undergo an MBS study for dysphagia and silent aspiration.
Laryngomalacia is the most common laryngeal anomaly and is commonly associated with stridor in children, but the recurrent respiratory and/or feeding difficulties associated with this condition may pose a threat to the well-being of the affected child.
To describe the prevalence of aspiration in pediatric patients with laryngomalacia who present with recurrent respiratory issues and/or feeding difficulty.
Design, Setting, and Participants
This retrospective review of medical records involved 142 pediatric patients. These patients received a laryngomalacia diagnosis, presented with recurrent respiratory and/or feeding difficulties, and underwent a modified barium swallow (MBS) study at a tertiary referral children’s hospital from January 1, 2015, to December 31, 2015. Each patient was assessed for aspiration and swallowing discoordination. Data collection and analysis were performed from December 1, 2016, to September 30, 2017.
Main Outcomes and Measures
Demographic data, presenting symptoms, medical comorbidities, and feeding and dysphagia details were collected and analyzed. Speech-language pathologists reviewed all MBS reports to identify the presence and type of aspiration (ie, silent vs nonsilent) with different textures and consistencies of food and liquid.
A total of 395 patients were diagnosed with laryngomalacia in 2015. One hundred forty-two of these patients (35.9%) presented with recurrent respiratory issues and/or feeding difficulties and were referred for MBS study for further evaluation. Ninety-two (64.8%) were male, with a median (interquartile range) age at the time of MBS study of 7.9 (3.2-20.5) months. Among these patients, 128 (90.1%) had swallowing dysfunction documented during the MBS study. Aspiration was identified in 60 patients (42.3%), and silent aspiration was documented in 59 (98.3%) of these 60 patients. Epilepsy or seizures (risk difference [RD], 11%; 95% CI, 5%-17%), laryngeal cleft (RD, 8%; 95% CI, 3%-13%), and premature birth (RD, 15%; 95% CI, 5%-25%) were statistically significantly associated with abnormal MBS findings.
Conclusions and Relevance
Swallowing dysfunction and aspiration were commonly found in pediatric patients with laryngomalacia and recurrent feeding and/or respiratory issues; these children should undergo an MBS study for dysphagia and silent aspiration.
Laryngomalacia is the most common congenital anomaly of the larynx.1,2 This condition involves a collapse of supraglottic tissue from excess mucosa or reduced laryngeal tone. Diagnosis of laryngomalacia is confirmed by flexible laryngoscopy, which is frequently performed as an office procedure. Common features include redundant arytenoid mucosa and cartilages with inspiratory prolapse, posterior displacement of the epiglottis, shortened aryepiglottic folds, and a tightly curled, omega-shaped epiglottis.3 Inspiratory stridor is the primary feature and commonly appears after the first 2 weeks of life.4 Surgical intervention is typically not required on initial presentation, and the pediatric patient’s progress is periodically monitored. In most cases, stridor and other symptoms of laryngomalacia will resolve by 2 years of age,5,6 and only 10% of patients require surgical intervention.4,7
Laryngomalacia is the most common cause of stridor in children, affecting up to 75% of stridulous infants in the United States.2,5,8 Laryngomalacia is also associated with a number of symptoms, particularly pertaining to feeding difficulties. Feeding difficulties, such as coughing, choking, gagging, regurgitation, emesis, and slow and/or inefficient feeding, may present in patients with laryngomalacia because of disruption of the suck-swallow-breathe sequence and airway protection.4 In addition to prolapsing supra-arytenoid tissue interfering with swallowing mechanisms, the laryngeal adductor reflex, which stimulates laryngeal elevation, epiglottic tilt, vocal fold closure, and cessation of inhalation as food passes into the esophagus, may be impaired in patients with laryngomalacia.9 Several reports suggest that neuromuscular impairment is the principal cause of laryngomalacia.4,5,10,11 The sensorimotor integrative function of the larynx not only explains why children with neurologic deficits tend to have more severe feeding difficulties and increased episodes of aspirations or silent aspiration but may also contribute to the prevalence of dysphagia in patients with laryngomalacia.12
Respiratory symptoms, such as recurrent respiratory infections or pneumonias, in infants with laryngomalacia may be caused or compounded by aspiration of fluid into the airway. Laryngeal penetration and aspiration are common findings in children with laryngomalacia,3,9 but the true prevalence has not been well established.3 Preoperative assessment for aspiration is not routinely performed, and mild cases of laryngomalacia without apparent symptoms of aspiration may remain undetected and undiagnosed. Silent aspiration, in which food or liquid pass into the airway below the vocal folds without stimulating a cough reflex, is of particular concern because of the lack of overt symptoms or clinical signs and the potential for chronic airway disease, including bronchiectasis, if left untreated. The true prevalence of silent aspiration is difficult to measure because of a lack of reliable and easily testable markers,13 but this condition has been shown to have a high prevalence in children with neurologic impairment,14 which raises the suspicion that the proposed neuromuscular origin of laryngomalacia may similarly contribute to silent aspiration.
Medical comorbidities may also predispose patients with laryngomalacia to aspiration or feeding difficulties, although findings to date have yielded conflicting results. A previous study suggested that gastroesophageal reflux disease (GERD), cardiac disease, and genetic or congenital anomalies may be associated with persistent aspiration after supraglottoplasty.9 However, a more recent study found that the presence of comorbidities, such as GERD, Down syndrome, neurologic impairment, or congenital heart disease, was not associated with swallowing dysfunction.3
The objective of the present study is to provide a snapshot of clinical characteristics of children with laryngomalacia who presented with recurrent respiratory issues and/or feeding difficulty and were referred for swallow assessment over a year-long period at our institution. We characterize dysphagia and aspiration and identify comorbidities that are associated with swallowing issues in this subset of pediatric patients. To our knowledge, no previous studies have specifically described the rates of silent aspiration in this patient population.
This retrospective review was approved by the Boston Children's Hospital Institutional Review Board. Patient informed consent was waived by this institutional review board because it determined that the study posed no more than minimal risk to the patients involved. Data collection and analysis were performed from December 1, 2016, to September 30, 2017.
We conducted an assessment for aspiration and swallowing discoordination in 142 pediatric patients with laryngomalacia who underwent at least 1 modified barium swallow (MBS) study at a tertiary referral hospital (Boston Children’s Hospital) from January 1, 2015, to December 31, 2015. All patients received a laryngomalacia diagnosis through flexible laryngoscopy or direct laryngoscopy and bronchoscopy and were evaluated for feeding difficulties and clinical signs of aspiration by the treating otolaryngologist (K.W., S.C., or R.R.) and a speech-language pathologist (J.P., P.D., K.H., or K.D.). The finding of aspiration was confirmed by an MBS study.
Complete medical records, including demographic information, operative reports, and MBS findings, were obtained and reviewed. MBS studies were performed in the radiology department by a speech-language pathologist (J.P., P.D., K.H., or K.D.) and a radiologist. Patients were offered various liquid consistencies of barium and/or pureed and solid foods mixed with barium. The presence and type of aspiration with different textures of food material were recorded and coded by speech-language pathologists (J.P., P.D., K.H., and K.D.) using the Penetration-Aspiration Scale (PAS; score range: 1-8, with the highest score indicating silent aspiration; Table 1). Patients were categorized along the scale according to their normal swallowing function (PAS 1), laryngeal penetration only (PAS 2-5), aspiration with protective response (PAS 6-7), or silent aspiration (PAS 8). The Functional Oral Intake Scale (FOIS)–Suckle Feeds and Transitional Feeds (Table 2) was used to characterize any resultant feeding impairment (eg, need for tube feeds and/or modified fluid or food texture). Swallowing dysfunction was reported if the patient had laryngeal penetration or aspiration, required special feeding equipment or a modified feeding regimen (ie, thickened fluids), or had a score of lower than 6 points on FOIS (score range: 1-6, with the highest score indicating a total oral diet with no restrictions).
The information we analyzed included demographic data, age at diagnosis of laryngomalacia, presenting symptoms, medical comorbidities, feeding difficulties, and aspiration and swallowing discoordination details. Characteristics of patients with laryngomalacia grouped by MBS study findings were compared using χ2 or Fisher exact test for categorical variables and Wilcoxon rank sum test for continuous variables. All statistical analyses were performed using SAS, version 9.4 (SAS Institute Inc).
Of the 395 pediatric patients who received a laryngomalacia diagnosis at our institution in 2015, 142 patients (35.9%) underwent an MBS study for recurrent respiratory issues (9 of 142 [6.3%]), feeding difficulty (98 [69.0%]), or both (35 [24.6%]). Of the 142 patients, 92 (64.8%) were male and 50 (35.2%) were female, with a median (interquartile range [IQR]) age at the time of MBS study of 7.9 (3.2-20.5) months (Table 3). Eighty-four patients (59.2%) had a GERD diagnosis, 59 (41.5%) had neurologic impairments, and 22 (20.0%) had a history of prematurity. In addition, 8 patients (5.6%) had laryngeal cleft, and 1 (0.7%) had unilateral vocal fold paralysis.
Fourteen patients (9.9%) exhibited normal swallowing on MBS study, and swallowing dysfunction was documented in 128 patients (90.1%). One hundred patients (70.4%) had abnormal MBS findings, defined as laryngeal penetration (40 [28.2%]) or aspiration (60 [42.3%]). Silent aspiration was demonstrated in 59 of the 60 patients with aspiration, making up 41.5% of the 142 patients with laryngomalacia who underwent MBS study and 14.9% of all 395 patients with laryngomalacia.
Patients who had abnormal MBS findings were compared with patients who had normal findings (Table 4). Epilepsy or seizures (risk difference [RD], 11%; 95% CI, 5%-17%), laryngeal cleft (RD, 8%; 95% CI, 3%-13%), and premature birth (RD, 15%; 95% CI, 5%-25%) were statistically significantly associated with abnormal MBS findings. No statistically significant differences were found in any other characteristics.
Children with laryngomalacia may have anatomical or neuromuscular disruption of mechanisms that protect the airway during feeding. This disruption explains some of the common signs and symptoms of laryngomalacia, such as coughing, choking, perioral pallor, cyanosis, apnea, and wheezing during feeding times.5,9,15,16 Infants with stridor who do not have substantial feeding-associated symptoms may be periodically monitored without surgical intervention. However, supraglottic airway obstruction caused by laryngomalacia may increase the work of breathing, thereby increasing infants’ energy expenditure. When combined with feeding disorders that lower energy intake, symptoms of laryngomalacia may lead to poor weight gain or even muscle atrophy.17 In addition, aspiration may further complicate existing respiratory issues, compounding the growth-faltering aspect of laryngomalacia. Thus, patients with symptoms of aspiration, worsening stridor, failure to thrive, and complications caused by airway obstruction and hypoxia may require supraglottoplasty, an endoscopic procedure that divides the aryepiglottic folds and excises arytenoid mucosa and/or cartilage to reduce collapse and prolapse into the airway.
Evidence has shown that laryngeal penetration and aspiration are particularly common in children with laryngomalacia. A previous study showed that among 50 infants with severe laryngomalacia who underwent supraglottoplasty, 36 (72%) had preoperative aspiration on a fiberoptic endoscopic evaluation of swallowing and 44 (88%) had laryngeal penetration.9 Moreover, 36 (82%) of the 44 patients with preoperative laryngeal penetration and 31 (86%) of the 36 patients with preoperative aspiration were reported to have resolution of these findings on postoperative fiberoptic endoscopic evaluation of swallowing. Richter et al9 suggest that, consistent with the neuromuscular theory of laryngomalacia, supraglottoplasty exposes neural endings in the densest area of superior laryngeal nerve fibers (ie, the aryepiglottic folds and supra-arytenoid tissue), thereby enhancing laryngeal sensation and airway protection. This procedure, in combination with the natural laryngeal maturation in infants with laryngomalacia,5 may reduce aspiration. However, note that altering the anatomy of the larynx through surgical intervention may also exacerbate aspiration. One report demonstrated transient postsupraglottoplasty swallowing dysfunction in 25% of children with laryngomalacia and a 17% rate of postsupraglottoplasty aspiration.7
This study identified swallowing dysfunction in more than 90% of patients with laryngomalacia who exhibited signs or symptoms concerning for aspiration and were referred for MBS study. Of these patients, those with epilepsy or seizures, laryngeal cleft, or a history of prematurity were statistically significantly more likely to have abnormal MBS findings. Other notable prevalent comorbidities included GERD, developmental delay, and congenital heart disease. In addition to this high prevalence of swallowing dysfunction, the results demonstrated a strong association between laryngomalacia and silent aspiration. Of the 142 patients we reviewed, 60 (42.3%) had aspiration, and 59 (98.3%) of these patients displayed silent aspiration (ie, no cough to clear). These data indicate that silent aspiration is present in 41.5% of patients with laryngomalacia who underwent MBS study or in 14.9% of the total number of patients with laryngomalacia. However, our findings are limited by the fact that the prevalence of silent aspiration was only measurable in patients who had an MBS study. The higher figure likely overestimates the prevalence of silent aspiration given that these patients were referred for MBS study because of symptoms suggestive of aspiration, whereas the lower figure may underestimate the prevalence as 253 patients (64.1%) were never formally evaluated for silent aspiration. We must assume that the true prevalence of silent aspiration in children with laryngomalacia lies somewhere between 14.9% and 41.5%.
The apparent tendency toward silent aspiration over non–silent aspiration further supports the need for comprehensive swallowing evaluations, including MBS, in the laryngomalacia patient population. The MBS study is considered the most reliable method for detecting silent aspiration, which may go unnoticed on a clinical feeding examination. Patients with laryngomalacia and recurrent respiratory issues that suggest the possibility of underlying swallowing dysfunction, such as acute respiratory illness or pneumonia, should be considered for an MBS study to evaluate for aspiration, even in the absence of overt coughing, choking, or other symptoms during feeding.
Identifying silent aspiration early in life may help prevent future associated morbidity. We recommend a multidisciplinary approach in the management of laryngomalacia, particularly in patients with medical comorbidities, involving a speech-language pathologist, an otolaryngologist, a pulmonologist, and a general pediatrician. Nasogastric feeding or gastrostomy should be considered if no safe oral diet is identified or if nutritional status is compromised. A neurologist may also play an important role in treating patients with neurologic comorbidities or patients who have persistent symptoms despite surgical intervention. It is recommended that a multidisciplinary team of clinicians be involved in the counseling of families and caregivers. Such counseling would help in the appropriate treatment of children with laryngomalacia and in the decision on whether supraglottoplasty or more conservative management is warranted.
This study is limited by its retrospective design. All MBS studies were performed in 2015, and only 1 was included per patient, with some serving as a patient’s first MBS study. Data collection as well as PAS and FOIS scoring were reliant on the MBS findings reported by the speech-language pathologist who conducted the feeding evaluation. Therefore, this study used only the data included in the MBS report. Another limitation was a possible selection bias because we reviewed the medical records of only those patients (among the many with laryngomalacia) who underwent an MBS study. Because these patients were referred for MBS study on the basis of their clinical presentation, they may not be completely representative of the entire population of children with laryngomalacia.
Future research should evaluate aspiration and dysphagia in children with laryngomalacia over a longer period, capturing data from preoperative, postoperative, and follow-up time points. A prospective longitudinal study of patients with laryngomalacia that assesses these data would help reveal the true prevalence of recurrent respiratory issues and/or feeding difficulty and aspiration in this patient population.
Most patients with laryngomalacia can be monitored conservatively. However, swallowing discoordination and aspiration deserve special consideration in patients with laryngomalacia who present with recurrent respiratory issues and/or feeding difficulty. Management may consist of diet modification, feeding strategies, counseling of families and caregivers, and supraglottoplasty.
Accepted for Publication: October 15, 2018.
Corresponding Author: Reza Rahbar, DMD, MD, Department of Otolaryngology and Communication Enhancement, Boston Children’s Hospital, 300 Longwood Ave, Boston, MA 02115 (firstname.lastname@example.org).
Published Online: December 27, 2018. doi:10.1001/jamaoto.2018.3642
Author Contributions: Dr Rahbar had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis. All authors contributed equally to this work.
Concept and design: Perez, Dodrill, Hernandez, Davidson, Rahbar.
Acquisition, analysis, or interpretation of data: Irace, Dombrowski, Kawai, Watters, Choi, Perez, Dodrill, Davidson.
Drafting of the manuscript: Irace, Perez.
Critical revision of the manuscript for important intellectual content: Irace, Dombrowski, Kawai, Watters, Choi, Dodrill, Hernandez, Davidson, Rahbar.
Statistical analysis: Irace, Dombrowski, Kawai.
Administrative, technical, or material support: Irace, Dombrowski, Dodrill, Davidson.
Supervision: Watters, Choi, Rahbar.
Conflict of Interest Disclosures: None reported.
Disclaimer: Dr Choi is deputy editor of JAMA Otolaryngology–Head & Neck Surgery, but she was not involved in any of the decisions regarding review of the manuscript or its acceptance.
Meeting Presentation: The results of this study were presented at the American Society of Pediatric Otolaryngology Annual Meeting; April 21, 2018; National Harbor, Maryland.
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