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OBSERVATIONS Dysfunction can exist at any of the 4 phases of the physiologic swallow.
Dysphagia manifests differently in children at each age in their development. Dysphagia
can present in otherwise healthy children but is more common in patients with a history of Author Affiliations: Department of
prematurity, neuromuscular disorders, cardiopulmonary disorders, anatomic anomalies of Otolaryngology, Children’s National
Health System, Washington, DC
the upper aerodigestive tract, and gastrointestinal tract disorders. Workup involves clinical
(Lawlor); Department of
feeding evaluations, imaging studies, and endoscopic evaluations. Appropriate management Otolaryngology & Communication
depends on the cause of dysphagia. Enhancement, Boston Children’s
Hospital, Boston, Massachusetts
CONCLUSIONS AND RELEVANCE The causes of dysphagia in children are varied and often (Choi).
multifactorial. Evaluation by a multidisciplinary team can facilitate accurate diagnosis and Corresponding Author: Sukgi Choi,
guide management. MD, MBA, Department of
Otolaryngology & Communication
Enhancement, Boston Children’s
JAMA Otolaryngol Head Neck Surg. doi:10.1001/jamaoto.2019.3622 Hospital, 300 Longwood Ave
Published online November 27, 2019. BCH3129, Boston, MA 02115 (sukgi.
choi@childrens.harvard.edu).
A
ppropriate nutritional intake is essential for the rapid phase; in fact, while eating, the respiratory rate becomes faster
growth and development that occurs during infancy and and more irregular than during tidal breathing.7,8 The esophageal
childhood. 1 Feeding and swallowing dysfunction are phase consists of cricopharyngeus relaxation, allowing the food
diagnosed with increasing frequency, especially in children with a bolus to enter the esophagus, and coordinated smooth muscle
history of prematurity, neuromuscular disorders, cardiopulmonary peristalsis passes the bolus into the stomach.2,9
disorders, anatomic anomalies of the upper aerodigestive tract, and Development of the suck and swallow begins in utero as
gastrointestinal tract disorders.2-4 Early diagnosis and intervention early as gestational week 10 or 11. Gestational age 34 to 38 weeks is
by a multidisciplinary team are essential to the management of typically when most children develop efficiency and tolerance of
swallowing disorders in children.5 oral feeding.5,10,11 In infants, all 4 phases are under involuntary re-
flex control. In children and adults, the preparatory and oral phases
Physiologic Characteristics of Swallowing are under voluntary control, and the pharyngeal and esophageal
The normal swallow is classically divided into 4 phases: the prepa- phases remain involuntary.2
ratory phase, the oral phase, the pharyngeal phase, and the A physiologic swallow is the result of the complex integration
esophageal phase. The preparatory phase is when food is taken of more than 30 nerves and muscles and must progress with the
into the oral cavity, moistened with saliva, chewed, and prepared child as their anatomy matures.2 Dysphagia is defined as difficulty
into a bolus using the oral tongue and hard palate. This phase swallowing and must be distinguished from behavioral feeding
develops at approximately age 6 months. Before age 6 months, disorders, such as oral aversion.1 Dysphagia can be further catego-
the preparatory phase consists of sucking from a nipple. The oral rized depending on the disordered phase of swallowing. Oral dys-
phase is the propulsion of the food bolus into the oropharynx by phagia can present as absent oral reflexes, immature or absent suck,
the oral tongue and the triggering of the swallow reflex. The soft uncoordinated biting/chewing, and poor handling of the food
palate elevates to prevent food from regurgitating into the naso- bolus. Pharyngeal dysphagia can present as laryngeal penetration,
pharynx. The pharyngeal phase is the passage of the food bolus when the food bolus enters the laryngeal vestibule; aspiration, when
through the oropharynx and hypopharynx toward the esophagus the food enters the airway below the vocal folds; choking, when food
via coordinated muscle contraction. The velum approximates the obstructs the airway; pharyngeal reflux; and nasopharyngeal reflux.1
pharyngeal musculature, the larynx elevates and the vocal folds Esophageal dysphagia results from obstruction of passage of the
adduct, and the tongue and pharyngeal muscles propel the bolus food bolus through the esophagus or by poor coordination of esoph-
into the pharynx.2,5,6 Respiration ceases during the pharyngeal ageal muscle contractions.
jamaotolaryngology.com (Reprinted) JAMA Otolaryngology–Head & Neck Surgery Published online November 27, 2019 E1
Epidemiologic Characteristics
The incidence of pediatric dysphagia is estimated to be 0.9% but Box. Disorders Predisposing Pediatric Patients to Swallowing
is thought to be higher in at-risk populations.2,12,13 As our ability to Dysfunction and Dysphagia
care for patients with extreme prematurity and complex medical
conditions, such as cerebral palsy, bronchopulmonary dysplasia, Structural Abnormalities
Nose and Nasopharynx
and cardiac anomalies improves, the incidence of swallowing dis-
Choanal stenosis or atresia
orders, increases.5 In addition, as many as 50% of parents report
Piriform aperture stenosis
that their otherwise healthy children have feeding problems and
as many as 80% of children with developmental delays may have Congenital intranasal masses
difficulties feeding.4 Midface hypoplasia
Inferior turbinate hypertrophy
Causes of Dysphagia Adenoid hypertrophy
The cause of dysphagia can result from a single medical issue but Nasopharyngeal masses
is commonly multifactorial, resulting from a confluence of comor-
bid conditions. Common causes of swallowing dysfunction can be Oral Cavity and Oropharynx
Oral ties
found in the Box. Determining the cause of dysphagia is vital to
identifying the appropriate intervention. Micrognathia or retrognathia
Cleft lip or palate
Prematurity Macroglossia
Prematurity is a common cause of dysphagia in infants.5,11,14-16 High arched palate
A large, population-based cohort study found the prevalence Congenital oral masses
of feeding problems in premature infants born at less than Tongue base masses
37 weeks’ gestation to be 10.5%, increasing to 24.5% among
those born with a very low birth weight of less than 1500 g.17 Hypopharynx and Larynx
Laryngomalacia
Delay in development of head control, tongue movement,
palatal function, gag reflex, and laryngeal sensation can Vocal fold immobility
contribute to poor swallow. 3 Premature infants often have Laryngotracheoesophageal cleft
comorbid pulmonary and central nervous system disease, further Glottic stenosis
complicating the dysphagia. Abnormalities of the aerodigestive Subglottic stenosis
system, such as laryngomalacia, or iatrogenic injury resulting Laryngeal masses
from respiratory support, such as prolonged intubation or
Vascular malformations
tracheostomy, can further complicate and delay development of a
normal swallow.3,18 Trachea and Esophagus
Esophageal atresia
E2 JAMA Otolaryngology–Head & Neck Surgery Published online November 27, 2019 (Reprinted) jamaotolaryngology.com
jamaotolaryngology.com (Reprinted) JAMA Otolaryngology–Head & Neck Surgery Published online November 27, 2019 E3
Figure 1. Algorithm for Evaluation and Management Figure 2. Image of Fluoroscopic Swallow Study
of Pediatric Dysphagia
Feeding difficulty
E4 JAMA Otolaryngology–Head & Neck Surgery Published online November 27, 2019 (Reprinted) jamaotolaryngology.com
and can be observed penetrating the airway during swallow. Ben- neurologists, otolaryngologists, pulmonologists, gastroenterolo-
efits of this examination include the ability to evaluate the breast- gists, dieticians, and SLPs.2,4 Neurologic evaluation is especially
feeding child and the lack of exposure to any radiation. Multiple evalu- important in children with dysphagia when no other abnormalities
ations of complete swallows can be performed without increased are found on comprehensive workup, including endoscopies. As with
risk to the patient. Limitations of flexible endoscopic evaluation of most pediatric illnesses, the caregiver is an important member of the
swallowing include tolerance of the nasopharyngoscopy, inability to treatment team. The key to successful management of dysphagia
view the oral phase of swallow, “white out” during swallow where is correctly identifying the cause.3
tissues contract that may obscure that examination, inability to de- Feeding therapy, performed by an experienced SLP, is often the
tect microaspiration, and the subjective nature of the evaluation.1,3-5 first-line treatment for infants and children with dysphagia. This
therapy may consist of altering the means of food delivery, includ-
Upper Gastrointestinal Tract Series ing the nipple flow or spoon, changing the feeding position, or pac-
The upper gastrointestinal tract series is a radiographic study that ex- ing the feeds in attempt to improve the suck-swallow-breathe
amines the esophagus, stomach, and duodenum during swallow of sequence.2,5,41 Sensory and motor exercises, also implemented by
barium-impregnatedfoodbolus.Thisstudycanbeusedtoevaluatethe a trained SLP, improve strength, movement, and coordination of the
esophagealphaseofswallowaswellastheanatomyofthesestructures.5 lips, tongue, jaw, soft palate, and pharyngeal muscles.4 Feeding
therapy should be attempted only if the child is deemed safe to
Imaging receive a trial of oral intake.
In children with concerns for recurrent aspiration or pneumonia, chest Different formulas may also be used in a trial to assess if they
radiography may be an important part of the workup. Chest radiog- are better tolerated by the patient.3 If a child is able to take some
raphy may demonstrate evidence of pneumonia or chronic lung dis- oral feeds, increasing the caloric value of feeds may improve
ease secondary to aspiration. Computed tomographic or magnetic nutrition.4 Consulting a pediatric dietician may be necessary to
resonance imaging of the head, neck, and/or chest may also be indi- ensure adequate nutritional intake. If changing the formula is not
cated to further evaluate suspected abnormal anatomy and for pre- successful, the consistencies of the food bolus may need to be
operative planning, such as for suspected choanal atresia, microgna- thickened. Thickened feeds may help to reduce or resolve laryn-
thia, and vascular anomalies. Computed tomographic imaging of the geal penetration, aspiration, and GERD. Thickeners have also been
chest can also demonstrate long-term sequelae of chronic aspira- shown to change the swallowing mechanics and improve pacing
tion, including bronchiectasis, air trapping, and bronchial thickening.3 during feeds by slowing bolus transit and improving bolus cohe-
sion during swallow.50 A variety of thickeners exist, including rice
Endoscopic Evaluations cereals, carob bean, and xanthum gum thickeners are on the mar-
Direct laryngoscopy, bronchoscopy, and esophagoscopies are ket and must be chosen carefully based on the patient’s age and
important to the complete evaluation of a patient with pharyngeal or comorbidities.2,50 Reflux regimens, including proton pump inhibi-
esophageal dysphagia and suspected penetration and/or aspiration. tors and histamine H2 receptor antagonists are often prescribed
Endoscopies may not be indicated in isolated oral dysphagia. The based on clinical symptoms of GERD. The use of acid suppression
examinations can often be performed during the same procedure in in children is controversial and may be associated with increased
the operating room with coordination by the otolaryngology, pulmo- hospitalizations. Many symptoms of dysphagia overlap with those
nology, and gastroenterology teams. Endoscopic evaluation pro- of GERD, resulting in inappropriate implementation of
vides the clinicians with an unparalleled examination of the patient’s pharmacotherapy.51,52 With time and conservative management,
anatomy. Direct laryngoscopy allows the otolaryngologist to assess many infants with aspiration will improve within 1 to 2 years.53
for anatomic anomalies of the oral cavity, oropharynx, hypopharynx,
supraglottis, glottis, and subglottis. Airway disorders, such as vallecu- Surgical Management
lar cyst, laryngomalacia, vocal fold immobility, and laryngotracheo- Surgical management of dysphagia in children is indicated when an
esophageal cleft, can be diagnosed.24-27 Tracheoscopy can diagnose anatomic abnormality is identified as the cause of the dysphagia. We
tracheomalacia, tracheal stenosis, and tracheoesophageal fistula.29 will focus on a few common causes relevant to the otolaryngologist.
Bronchoscopymayassessforbronchomalacia,bronchiectasis,andevi-
dence of chronic aspiration and pneumonias. Additional testing, such Ankyloglossia
as biopsies, bronchial washing, pepsin, and lipid-laden macrophage Ankyloglossia is an area of great interest and controversy within the
indices, can supplement the endoscopy.47-49 Esophagoscopy af- feeding literature. Ankyloglossia has been identified as a cause of
fords an examination of the esophagus and, sometimes, distal struc- shallow latch, poor oral seal, and maternal nipple pain.20 Fre-
tures, including the stomach and duodenum. Extrinsic compression notomy is the division in the lingual frenulum. A variety of tech-
and mucosal changes can be visualized. Biopsies may be performed niques have been described, the simplest of which involves using
to evaluate for eosinophilic esophagitis and complications of GERD.30 a scissor to incise the frenulum without anesthesia, cautery, or
A multichannel, intraluminal impedance probe may be placed to sutures in young infants. Frenotomy improves feeding in many pa-
assess for acid and nonacid reflux.2 tients with ankyloglossia and restricted tongue range of motion. It
is generally a low-risk procedure, with infrequent complications in-
Medical Management cluding bleeding, infection, injury to the salivary ducts, and need for
Children with dysphagia benefit from the care of a multidisci- revision. However, there is growing concern that ankyloglossia is
plinary team that specializes in pediatric swallowing disorders. This overdiagnosed and treated. A tight lingual frenulum is often iden-
team may consist of pediatricians, developmental pediatricians, tified as the cause of difficulty with breastfeeding, when in fact the
jamaotolaryngology.com (Reprinted) JAMA Otolaryngology–Head & Neck Surgery Published online November 27, 2019 E5
difficulty is often multifactorial, relating to mandible position, ma- toplasty has been shown to improve swallow outcomes in children
ternal nipple, oral coordination, and other factors.20,54,55 Pub- with laryngomalacia.56-59 However, supraglottoplasty can result in
lished data on frenotomy are limited by poor standardization of new-onset aspiration in patients without previous swallow
diagnosis of ankyloglossia and lack of validated outcomes. To our dysfunction.60,61 Children with baseline neurologic dysfunction
knowledge, there is no literature to support the surgical manage- and a history of prematurity have an increased risk of aspiration
ment of other oral ties, such as upper lip, lower lip, or lateral ties.20 following supraglottoplasty.62,63
E6 JAMA Otolaryngology–Head & Neck Surgery Published online November 27, 2019 (Reprinted) jamaotolaryngology.com
patients without swallowing dysfunction when direct laryngoscopy vocal fold immobility include injection medialization laryngo-
is performed for other indications, so the anatomic finding alone is plasty, thyroplasty, and reinnervation procedures.70,71 Proper
not a reason to intervene. In patients with type 1 laryngeal cleft and procedural selection is based on the patient’s age, prognosis
dysphagia or aspiration, most surgeons advocate for a period of for recovery of vocal fold function, and the overall patient condi-
conservative medical management with thickened feeds ranging tion. The most common procedure performed for dysphagia
from 3 to 12 months.66 Surgical repair of type 1 laryngeal clefts con- and vocal fold immobility is injection medialization laryngoplasty
sists of interarytenoid injection augmentation with a filler agent (Figure 4), which is approached via suspension microlaryn-
(carboxymethylcellulose, fat, hyaluronic acid, gelfoam) or inter- goscopy. Common injectates include carboxymethylcellulose,
arytenoid suture repair with sharp dissection or the use of laser calcium hydroxyapatite, micronized acellular dermis, and
(Figure 3).64,66,67 Both procedures are performed endoscopically via hyaluronic acid; each has a variable duration of benefit. 70,71
suspension microlaryngoscopy. Injection and suture repair have The site of injection is the junction of the medial third and the
both resulted in improved swallow outcomes in children with type 1 posterior third of the vocal fold. The injectate is applied within
laryngeal cleft, although dysphagia and aspiration can persist, espe- the paraglottic space and the vocal fold is overmedialized by
cially if the cause was initially multifactorial.64,65,67 Type 2 laryngeal approximately 20% to account for resorption. Early injection
clefts are typically approached endoscopically. Types 3 and 4 laryn- seems to result in greater benefit to the patient.71 In one study,
geal clefts are classically repaired via an open approach, although type injection medialization laryngoplasty resulted in objective
3 clefts have been repaired successfully using endoscopes.68 improvement in swallow in over 60% of the patients and allowed
for liberalization of diet.69
Vocal Fold Immobility
Vocal fold immobility in infants and children can be congenital or ac-
quired. The restricted mobility of 1 or both vocal cords can result in
Conclusions
insufficient cough, glottic incompetence, and frank or silent
aspiration.26,69 However, not all children with unilateral or bilateral Dysphagia is an increasingly common disorder in pediatric
vocal fold immobility have an unsafe swallow. In addition, depend- patients, especially as advances in health care increase the sur-
ing on the cause, varying degrees of vocal fold function may re- vival of premature and medically complex children. The causes of
cover as late as 18 months after injury.69 dysphagia are varied and often multifactorial. Evaluation by a
When both swallowing dysfunction and vocal fold immobility multidisciplinary team can facilitate accurate diagnosis and guide
are diagnosed, intervention is indicated. Surgical approaches to management.
ARTICLE INFORMATION source, and management. Otolaryngol Clin North Am. 12. Bhattacharyya N. The prevalence of pediatric
Accepted for Publication: October 6, 2019. 2014;47(5):691-720. doi:10.1016/j.otc.2014.06.004 voice and swallowing problems in the United
4. Borowitz KC, Borowitz SM. Feeding problems in States. Laryngoscope. 2015;125(3):746-750.
Published Online: November 27, 2019. doi:10.1002/lary.24931
doi:10.1001/jamaoto.2019.3622 infants and children: assessment and etiology.
Pediatr Clin North Am. 2018;65(1):59-72. doi:10. 13. Svystun O, Johannsen W, Persad R, Turner JM,
Author Contributions: Dr Choi had full access to all 1016/j.pcl.2017.08.021 Majaesic C, El-Hakim H. Dysphagia in healthy
of the data in the study and takes responsibility for children: characteristics and management of a
the integrity of the data and the accuracy of the 5. Kakodkar K, Schroeder JW Jr. Pediatric
dysphagia. Pediatr Clin North Am. 2013;60(4):969- consecutive cohort at a tertiary centre. Int J Pediatr
data analysis. Otorhinolaryngol. 2017;99:54-59. doi:10.1016/
Concept and design: Both authors. 977. doi:10.1016/j.pcl.2013.04.010
j.ijporl.2017.05.024
Acquisition, analysis, or interpretation of data: 6. Delaney AL, Arvedson JC. Development of
Lawlor. swallowing and feeding: prenatal through first year 14. Rommel N, De Meyer AM, Feenstra L,
Drafting of the manuscript: Lawlor. of life. Dev Disabil Res Rev. 2008;14(2):105-117. Veereman-Wauters G. The complexity of feeding
Critical revision of the manuscript for important doi:10.1002/ddrr.16 problems in 700 infants and young children
intellectual content: Both authors. presenting to a tertiary care institution. J Pediatr
7. McFarland DH, Lund JP. Modification of Gastroenterol Nutr. 2003;37(1):75-84. doi:10.1097/
Supervision: Choi. mastication and respiration during swallowing in 00005176-200307000-00014
Conflict of Interest Disclosures: No disclosures the adult human. J Neurophysiol. 1995;74(4):1509-
were reported. 1517. doi:10.1152/jn.1995.74.4.1509 15. Rommel N, Bellon E, Hermans R, et al.
Development of the orohypopharyngeal cavity in
Disclaimer: Dr Choi is a deputy editor of JAMA 8. Derkay CS, Schechter GL. Anatomy and normal infants and young children. Cleft Palate
Otolaryngology–Head & Neck Surgery, but she was physiology of pediatric swallowing disorders. Craniofac J. 2003;40(6):606-611. doi:10.1597/1545-
not involved in any of the decisions regarding Otolaryngol Clin North Am. 1998;31(3):397-404. 1569_2003_040_0606_dotoci_2.0.co_2
review of the manuscript or its acceptance. doi:10.1016/S0030-6665(05)70060-8
16. Seddon PC, Khan Y. Respiratory problems in
9. Miller CK, Willging JP. Advances in the evaluation children with neurological impairment. Arch Dis Child.
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