Dysphagia in Children: Gastroesophageal Causes

Central Annals of Otolaryngology and Rhinology

Review Article *Corresponding author

Stave Salgado Karen, Pediatric doctor specialist in Child Gastroenterologist. Ex-scholarship of the Dysphagia in Children: Gastroenterology Service of Hospitalde Pediatría S.A.M.I.C. Prof. Dr. Juan P. Garrahan, Buenos Aires – Maipú 863, CP 1006, Buenos Aires, Argentina, Tel: +54 Gastroesophageal Causes 1126387622; E-mail: Stave Salgado Karen Vanessa* Submitted: 12 March 2018 Accepted: 05 April 2018 Pediatric doctor specialist in Child Gastroenterologist, Ex-scholarship of the Published: 06 April 2018 Gastroenterology Service of Hospitalde Pediatría, Argentina ISSN: 2379-948X Copyright Abstract © 2018 Karen Vanessa Dysphagia is any disruption to the swallow sequence that results in compromise to the safety, efficiency, or adequacy of nutritional intake. Pediatric dysphagia OPEN ACCESS has focused largely on a number of specific populations at risk for swallowing difficulties, such as children with cerebral palsy, acquired/traumatic brain injury, other Keywords neuromuscular disorders, craniofacial malformations, airway malformations, congenital • Dysphagia cardiac disease, and gastrointestinal disease. • Swallowing disorders • Children The gastroesophageal disorders that cause dysphagia and swallowing/feeding • Pediatric dysphagia difficulties, such as gastroesophageal gastroesophageal reflux disease, eosinophilic esophagitis and achalasia need an early diagnosis because they can compromise quality of life and produce complications at the patients.

ABBREVIATIONS UES: Upper Esophageal Sphincter; OA: Oesophageal Atresia; pharyngeal an esophageal phase. In the first phase, also known TEF: Tracheo-Oesophageal Fistula; GER: Gastroesophageal as the oral preparatory phase, the food is taken into the oral cavity, chewed and moistened with saliva, and prepared into a Esophagitis; LES: Lower Esophageal Sphincter; VFSS: Reflux; GERD: Gastroesophageal Reflux Disease; Eoe: Eosinophilic bolus, which is held between the hard palate and oral tongue. This process first becomes evident at approximately 6 months Evaluation of Swallow; UGI: Upper Gastrointestinal Series. Videofluoroscopic Swallow Study; FEES: Fiberoptic Endoscopic of age [1]. The oral phase consists of salivation, mastication INTRODUCTION toand upper the transportation esophageal sphincter of the bolus (UES); towards and the the last pharynx. phase include In the pharyngeal phase the bolus is transported through the pharynx

Dysphagia refers to problems in any of phases of swallowing. transportation of the bolus through the esophagus to the stomach When referring to pediatric populations, the terms feeding [3]. and swallowing difficulties are more frequently used and are The sequence of events during the pharyngeal and becoming more common, particularly in infants born prematurely diagnostic categories associated with pediatric swallowing and esophageal phases remains the same throughout a person’s life, and in children with chronic medical conditions [1,2]. General and these events can be summarized as follows: (a) closure of the nasopharyngeal port through movement of the velum; (b) feeding difficulties include neurological conditions, anatomical pharyngeal closure through contraction of the superior, middle, anomalies, gastrointestinal disorders, conditions affecting and inferior pharyngeal constrictors; (c) closure of the vocal folds sucking swallowing and breathing coordination, and genetic with brief cessation of respiration; (d) hyolaryngeal excursion conditions [2]. and closure of the larynx through epiglottic tilt; (e) opening The consequences of dysphagia can be debilitating of the upper esophageal sphincter through relaxation of the for children, as it may lead to failure to thrive, respiratory cricopharyngeus muscle and biomechanical forces contributed complications, and compromised quality of life therefore early through hyolaryngeal excursion, and (f) peristaltic contraction of diagnosis is critical [1,2]. the esophagus to move the food or liquid into the stomach [3]. The objective of this clinical review is to provide a global In neonates and young infants, all components of swallowing understanding of the common gastroesophageal causes of are reflexive and involuntary. Later in infancy, the oral phase dysphagia in children in order to make a suitable diagnosis and comes under voluntary control, which is important to allow treatment.Normal swallowing childrenAbnormal to begin swallowing to masticate solid food.

The act of swallowing includes four stages: preparatory, oral, Any disruption to the swallow sequence is called dysphagia, Cite this article: Karen Vanessa SS (2018) Dysphagia in Children: Gastroesophageal Causes. Ann Otolaryngol Rhinol 5(2): 1208. Karen Vanessa (2018) Email: [email protected] Central of nutritional intake [3]. that results in compromise to the safety, efficiency, or adequacy etiology for dysphagia. Esophagography after OA repair should be performed because of the high index of suspicion for the presence of distal congenital esophageal stricture. Endoscopy Approximately 1% of children in the general population will with biopsies allows the evaluation of the anastomosis (stricture, experience swallowing disorders, though the incidence rate is diverticulum), the esophageal mucosa (peptic, eosinophilic or much higher in infants born prematurely and in children with infectious esophagitis) and the diagnosis of congenital stenosis, chronic medical conditions [4,5]. mucosal bridge, inlet patch or extrinsic compression (vascular It is important distinguish dysphagia as a skill-based disorder, anomalies, tight fundoplication wrap) (Figure 1) [10,11]. which is very different from a behaviorally based feeding Esophageal motility can be assessed by esophageal disorder. Behavioral feeding disturbances occur when a child manometry. The patterns of esophageal dysmotility in a cohort of is unwilling to consume a fluid/food despite sufficient physical children with OA were recently described using high resolution skills to do so. are [3]: manometry and were reported abnormal in all patients, with The common presentations of pediatric dysphagia symptoms 3 types of abnormalities observed: pressurization (15%), isolated distal contractions (50%) and a peristalsis (35%) [12]. The management of dysphagia must be conducted according • Oral phase: Absent oral reflexes, primitive/neurological to the underlying cause, such as feeding adaptation treatment oral reflexes, weak suck, uncoordinated suck, immature biting of esophagitis (peptic, eosinophilic or infectious), prokinetics, and/or chewing, disordered biting and/or chewing, poor bolus surgical repair of vascular anomaly, gastrostomy tube feeding or propulsion, poor bolus containment. dilationGastroesophageal of fundoplication reflux [10]. and gastroesophageal • Pharyngeal phase: Laryngeal penetration, aspiration, reflux disease choking,Causes pharyngealof dysphagia residue, nasopharyngeal reflux.

Gastroesophageal reflux (GER) is the passage of gastric The causes of dysphagia in pediatric populations are often contents into the esophagus with or without regurgitation and somewhat different than in adult patients, and children can ofvomiting. infants [7,13].Repeated expulsion of gastroesophageal contents from present with multiple variations of swallowing impairments the oral cavity in GER is reported to occur in approximately 40% [3,6,7].affecting any or all of the phases of swallowing. Table 1 summarizes common causes of dysphagia in pediatric patients Gastroesophageal causes of dysphagia Gastroesophageal reflux disease (GERD) is present when the reflux of gastric contents causes troublesome symptoms and/or Oesophageal atresia: complications. The prevalence of symptomatic or pathologic GER or GERD is estimated to occur in 10% to 20% of infants in North Oesophageal atresia (OA) is a America. Some children are at higher risk of GERD, including congenital malformation, characterized by an interruption those with neurologic impairment, obesity, esophageal achalasia, in the continuity of the oesophagus and have an incidence of hiatal hernia, prematurity, bronchopulmonary dysplasia, and OA. around 1:2500 live-births. It may be divided anatomically into GERD can contribute to persistent dysphagia by reducing mucosal 5 types with the most common being oesophageal atresia with sensation and laryngeal reactivity during the pharyngeal phase a distal tracheo-oesophageal fistula (TEF) found in around 85% of swallowing owing to mucosal injury by caustic reflex contents cases. The condition consists of a discontinuity or atresia of the [7,13]. Coppens et al, confirmed the association of dysphagia with oesophagus; with the majority of infants exhibiting a connection GERD, with or without fundoplication, in children with repaired or fistula between the oesophagus and trachea. The exceptions OA. This is in accordance with previous research, which indicates to this are children born with an isolated OA and those with an abnormal oesophageal motility as common etiologic factor [9]. H-type TEF [8,9]. In infants and toddlers, there is no symptom or symptom Dysphagia is a common problem in children with repaired complex that is diagnostic of GERD or predicts response to OA, with ranges between 45-70%. The etiology of dysphagia therapy. In older children and adolescents, as in adult patients, may include inflammatory or anatomic causes such as peptic history and physical examination may be sufficient to diagnose esophagitis, eosinophilic esophagitis, anastomotic stricture, GERD if the symptoms are typical. Complications of GERD congenital stenosis, peptic stricture, post-fundoplication are esophagitis, peptic stenosis and Barrett esophagus. The obstruction, vascular anomalies, anastomotic diverticulum, Combined Multiple Intraluminal Impedance and pH monitoring mucosal bridge, and inlet patch. In the absence of the latter detects acid, weakly acid, and nonacid reflux episodes. It is the causes, esophageal dysmotility remains the accepted explanation gold standard for GERD and is superior to pH monitoring alone [9,10]. for evaluation of the temporal relation between symptoms and Dysphagia can present with simply a complaint of difficulty GER. Esophageal manometry may be abnormal in patients with in swallowing (50%), nausea (27%), epigastric burning (21%), GERD but the findings arenot sufficiently sensitive or specific to heartburn (14%-50%), postprandial fullness (14%), early satiety confirm a diagnosis of GERD, nor to predict response to medical (14%), eructation (14%), regurgitation (7%-50%), or epigastric or surgical therapy. Endoscopically visible breaks in the distal pain (7%). Evaluation of dysphagia in OA patients should begin esophageal mucosa are the most reliable evidence of reflux with contrast studies that can be helpful in identifying a structural esophagitis. Endoscopic biopsy is important to identify or rule Ann Otolaryngol Rhinol 5(2): 1208 (2018) 2/5 Karen Vanessa (2018) Email: [email protected] Central

Table 1:

Prematurity Disorders commonly affectingCongenital feeding and abnormalities swallowing in children.Iatrogenic complications Gastrointestinal disorders

- Low gestational age at birth -Tongue tie -Tube feeding - OA/TEF prematurity- Low birth weight -Cleft lip/palate -Tracheostomy -GERD - Comorbidities associated with -Moebius syndrome -Respiratory support -EoE -Down syndrome arousal,-Certain medications -Food allergies and intolerances (especially those that affect -Achalasia -Congenital diaphragmatic hernia awareness, muscle tone, or -Necrotizing enterocolitis saliva production) -Hirschsprung disease Respiratory and cardiac Neuromuscular disorders Ingestional (caustic) Maternal and perinatal issues -Gastroschisis disorders injuries

-Microcephaly -Jaundice -Apnea -Hydrocephalus -Cleaning agents -Diabetes -Pulmonary dysplasia -Intraventricular hemorrhage -Battery -Fetal alcohol syndrome -Respiratory distress syndrome -Periventricularleukomalacia -Neonatal abstinence syndrome -Bronchopulmonary dysplasia -Cerebral palsy -Laryngo-/tracheo-/ -Seizures bronchomalacia -Muscular dystrophy Abbreviations: -Heart defects OA: Oesophageal Atresia; TEF: Tracheo-Oesophageal Fistula; GERD: Gastroesophageal Reflux Disease; EoE: Eosinophilic Esophagitis

pump inhibitors) and surgery with a Nissen fundoplication what may be performed on children at risk for life-threatening Eosinophiliccomplications of esophagitis GERD [14].

Eosinophilic esophagitis (EoE) is a chronic immune-mediated condition characterized by clinical symptoms secondary to esophageal dysfunction and histologically by eosinophilic infiltration of the esophagus [15]. Figure 1 Clinical symptoms vary according to age. Feeding difficulties (a) Barium swallow of a symptomatic EoE stricture in OA– are the most common symptoms in infants and toddlers (including TEF patient. (b) EoE stricture in EA-TEF patient as seen endoscopically vomiting, regurgitation and feeding refusal). During childhood, [Krishnan U. Eosinophilic Esophagitis in Children with Esophageal Atresia. Eur J Pediatr Surg. 2015; 25: 336-344]. vomiting and/or abdominal or retrosternal pain are reported, whereas during adolescence, GERD symptoms, dysphagia, and food impaction are the most frequent symptoms [7,15]. The consensus recommendation identified four dominant presenting symptoms of esophageal dysfunction: dysphagia, abdominal pain, GERD/vomiting, and failure to thrive/feeding difficulty [16].

Figure 2

Upper gastrointestinal study demonstrating a “bird’s beak” deformity (arrow) in a 17-year-old patient with achalasia. (Image provided by R. Ignacio 2016). out other causes of esophagitis, and to diagnose and monitor Barrett esophagus and its complications [13]. Figure 3 The management of GERD includes dietary and lifestyle changes, medications (Histamine 2 receptor antagonists, Proton Videofluoroscopy of a patient with dysphagia. Ann Otolaryngol Rhinol 5(2): 1208 (2018) 3/5 Karen Vanessa (2018) Email: [email protected] Central

Cricopharyngeal achalasia

The updated definition of the disease includes the histological presence of ≥15 eosinophils per high power field in at least The cricopharyngeus muscle is a striated muscle that is 1 endoscopic esophageal mucosal biopsy (peak value) taken contracted at rest, thus keeping the esophagus closed during at upper gastrointestinal endoscopy; and/or the presence of respiration. Cricopharyngeal achalasia is thought to involve other microscopic features of eosinophilic inflammation such as spasm or incomplete relaxation of the cricopharyngeus muscle. eosinophilic micro abscesses, superficial layering, or extracellular This is a rare cause of dysphagia in children and may develop eosinophil granules [15]. EoE patients with dysphagia have between birth to 6 months of age. However, diagnosis may be significantly higher eosinophils compared to EoE patients with delayed due to non-specific symptoms including choking, food abdominal pain, and the level of inflammation as seen from regurgitation, nasal reflux, coughing recurrent pneumonia, eosinophil micro abscesses, superficial layering, desquamation, cyanosis and failure to thrive [7, 22]. and the distribution around rete pegs is significantly higher [17]. This disorder can be diagnosed by identification of a prominent Besides, recent studies suggest an increased incidence of bar on videofluoroscopic swallow study (VFSS) and increased EoE in OA–TEF patients. As presenting symptoms of EoE are pressures proximal to the muscle can also be demonstrated on similar to those of GERD, misdiagnosis or delayed diagnosis often manometry (Figure 3). The use of endoscopic dilatation is the occurs in OA patients, in whom anastomotic strictures, GERD, first option because it is not an invasive technique, but it usually and dysphagia are common postoperatively[18,19]. Therefore, it requires several sessions. The second-line therapy is surgery, a is suggested that if these symptoms persist in patients with OA, more aggressive technique. In addition, the injection of botulinum performanupper digestive endoscopy with biopsies to rule out toxin represents a safe and effective alternative, although it is a Assessment techniques associated EoE [11]. less widespread method in pediatrics [7,23]. The management consists of dietary modification and reflux therapy. Three diet forms are commonly prescribed: an elemental Techniques used for diagnosing and monitoring pediatric diet that is a liquid formula based on amino acids and free of dysphagia include clinical evaluation tools and quality of life all allergens, a 6-food elimination diet that removes commonly measures, as well as a range of instrumental evaluation tools. identified allergens, or a targeted elimination diet that eliminates VFSS and fiberoptic endoscopic evaluation of swallow (FEES) are food identified as allergic to patient after testing. Swallowed the most commonly used instrumental assessments in pediatric corticosteroids are also effective in treating acute exacerbations dysphagia and they are considered gold standards in the ofAchalasia EoE but the disease often relapses after discontinuation [15]. diagnostic assessment of swallowing problems and aspiration [2,3] (Table). VFSS allows for the assessment of the swallow in all of the Achalasia is a rare esophageal neurodegenerative disorder swallowing stages. During this study, the patient is presented characterized by failure of lower esophageal sphincter (LES) with barium-impregnated liquid and food, and video fluoroscopic relaxation. The incidence of achalasia in childhood is 0.11/100000 monitoring is used to document oropharyngeal swallow function children annually. The pathophysiologic basis of achalasia is and swallowing disturbances [3]. In contrast to the VFSS, the characterized by the degeneration of the inhibitory my enteric FEES exam does not require intake of barium or radiation plexus that innervates the LES and esophageal body. This leads to exposure, but it does require that a patient tolerate the passing an imbalance in the inhibitory and excitatory neurons resulting of a nasal endoscope. FEES provides images of the larynx and in the failure of the LES to relax with swallowing, absence of hypopharynx before and after the pharyngeal swallow, which peristalsis of the esophageal body, and increased LES resting allows the detection of structural and physiological swallowing pressures [20]. impairments, as well as an assessment of aspiration risk [24]. Children usually present with progressive dysphagia, The upper gastrointestinal series (UGI) is a radiologic examination of the upper gastrointestinal tract and consists of a vomiting, and weight loss. Younger children and infants. may also present atypically with recurrent pneumonia, nocturnal cough, series of radiographic images delineating the esophagus, stomach, and duodenum. In the setting of dysphagia, a UGI can be helpful aspiration, hoarseness, and feeding difficulties [21] by noting anatomic and functional abnormalities, obstructions, Achalasia is diagnosed with a barium swallow study and as well as physiology of the oropharyngeal structures and UGI may be confirmed with esophageal manometry. Barium swallow system. Esophagoscopy allows assessment and, if necessary, studies classically demonstrate a dilated esophagus with “bird’s- biopsies to identify GERD and EoE [1,7]. beak” like tapering of the distal esophagus (Figure 2). Elevated Other tools have received recent attention for their resting LES pressure, absent or low-amplitude peristalsis, or diagnostic usefulness as adjunct assessments for the diagnosis non-relaxing LES upon swallowing are diagnostic findings of dysphagia in pediatric populations, such as manometry and on esophageal manometry in children with achalasia. The impedance. These tools provide information about pharyngeal various methods of treatment of achalasia involve reduction and esophageal motility, as well as presence of Gastroesophageal of LES pressure in order to facilitate esophageal emptying by: refluxMANAGEMENT [3]. oral administration of calcium channel blockers, pneumatic dilatation, injection of botulinum toxin, esophageal myotomy with or without an anti-reflux procedure, and a novel technique Therapy intervention for children with oral-phase swallowing called peroral endoscopic myotomy [20]. problems generally involves exercises aimed at improving the Ann Otolaryngol Rhinol 5(2): 1208 (2018) 4/5 Karen Vanessa (2018) Email: [email protected] Central

11. sensory and/or motor skills required for drinking and eating. Stave Salgado KV, Rocca AM. Esofagitis eosinofílica y atresia esofágica: For children with swallowing problems affecting the pharyngeal casualidad o causalidad. Arch Argent Pediatr. 2018; 116: 61-69. phase, therapy intervention generally involves the child to 12. Lemoine C, Aspirot A, Le Henaff G, Piloquet H, Lévesque D, Faure C. modify their swallowing strategy or teaching the feeder to modify Characterization of esophageal motility following esophageal atresia the bolus [3]. Furthermore, the treatment of dysphagia must be repair using high-resolution esophageal manometry. J Pediatr 13. Gastroenterol Nutr. 2013; 56: 609-614. conductedCONCLUSION according to the underlying cause. Vandenplas Y, Rudolph C, Di Lorenzo C, Hassall E, Liptak G, Mazur L, et al. Pediatric Gastroesophageal Reflux Clinical Practice Guidelines: Joint Recommendations of the North American Society for Pediatric Children have rapidly developing body systems and even Gastroenterology, Hepatology, and Nutrition (NASPGHAN) and the short-term problems with swallowing can interrupt normal European Society for Pediatric Gastroenterology, Hepatology, and development and cause serious long-term sequelae. Nutrition (ESPGHAN). J Pediatr Gastroenterol Nutr. 2009; 49: 498- 547. Populations at particular risk of dysphagia include children with cerebral palsy, neuromuscular disorders, craniofacial 14. Papachrisanth. Davis R. Clinical Practice Guidelines for the malformations, congenital cardiac disease, children born preterm Management of Gastroesophageal Reflux and Gastroesophageal and children with gastrointestinal disease. The gastroesophageal Reflux Disease: 1 Year to 18 Years of Age. J Pediatr Health Care. 2016; 30: 289-294. causes of dysphagia should be suspected in order to make an approximationREFERENCES to the diagnosis and proper management. 15. Papadopoulou A, Koletzko S, Heuschkel R, Dias JA, Allen KJ, Murch SH, et al. Management Guidelines of Eosinophilic Esophagitis in 1. 16. Childhood. J Pediatr Gastroenterol Nutr. 2014; 58: 107-118. Kakodkar K, Schroeder JW. Pediatric dysphagia. Pediatr Clin N Am. Liacouras CA, Furuta GT, Hirano I, Atkins D, Attwood SE, Bonis PA, et 2013; 60: 969-977. al. Eosinophilic esophagitis: updated consensus recommendations for 17. children and adults. J Allergy ClinI mmunol. 2011; 128: 3-20. 2. Speyer R, Cordier R, Parsons L, Denman D, Kim JH. Psychometric Characteristics of Non-Instrumental Swallowing and Feeding Gunasekaran T,Christopher C, Ronquillo N, Chennuri R, Adley Assessments in Pediatrics: A Systematic Review Using COSMIN. B, Borgen K, et al. Detailed Histologic Evaluation of Eosinophilic 3. Dysphagia. 2018; 33:1-14. Esophagitis in Pediatric Patients Presenting with Dysphagia or Dodrill P, Gosa M. Pediatric Dysphagia: Physiology, Assessment, and Abdominal Pain and Comparison of the Histology between the Two Management. Ann Nutr Metab. 2015; 66: 24-31. Groups. Can J Gastroenterol Hepatol. 2017; 2017: 3709254.

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Cite this article Karen Vanessa SS (2018) Dysphagia in Children: Gastroesophageal Causes. Ann Otolaryngol Rhinol 5(2): 1208.

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