A seven-month-old with chronic intermittent biphasic stridor

Abstract

Paediatric stridor can indicate serious pathology and requires prompt evaluation and management. Causes range from laryngomalacia to aerodigestive tract foreign bodies. We report on a chronic presentation of paediatric stridor, discuss the workup, management and lessons learned. A seven-month-old boy presented with chronic intermittent biphasic stridor for the previous five months. Diagnostic microlaryngoscopy and bronchoscopy revealed significant localised tracheomalacia. Urgent chest computed tomography was performed to investigate further. Radiological findings revealed an oesophageal foreign body prompting immediate rigid oesophagoscopy and removal of an embedded pistachio shell. The patient had an uneventful recovery. Foreign bodies in the airway and oesophagus are quite common in children, causing symptoms from the aerodigestive tract. It is uncommon for oesophageal foreign bodies to present with stridor in the absence of digestive tract symptoms. The otolaryngologist should consider this as a diagnosis, particularly in young children with an atypical presentation and symptoms.

Background

Stridor is the cardinal sign of airway obstruction and is a frequent presentation in the paediatric population. in children under one year of age, the most common cause is laryngomalacia, with foreign body inhalation becoming the most common cause from 1–12 years.¹ A good clinical history and examination with flexible laryngoscopy are usually sufficient in securing a diagnosis. In the absence of other clinical findings and in all but the mildest of cases, further investigation is often warranted in the form of diagnostic microlaryngoscopy and bronchoscopy to completely evaluate the airway for congenital structural abnormalities or acquired causes of airway stenosis. We present the case of a seven-month-old boy who presented with a history of stridor and recurrent croup, which was subsequently found to be due to an oesophageal foreign body causing external compression of the trachea.

Case history

A seven-month-old infant presented to the paediatric ear, nose and throat outpatient clinic with intermittent noisy breathing, stridor and chronic cough for five months, according to his parents. There was no preceding history of foreign body ingestion. Symptoms worsened when the patient was agitated, unwell or crying. Past medical history included 12 recurrent croup-like episodes which responded promptly to medical treatment provided in the emergency department with no requirement for intubation. No cyanosis was ever noticed. There were no feeding difficulties. He had a normal perinatal history with age appropriate growth and development. Except for the stridor, which was witnessed in clinic and characterised as being biphasic, clinical examination was normal.

Given the history, a diagnostic microlaryngoscopy and bronchoscopy were scheduled to exclude any congenital or acquired airway anomalies. Intraoperatively, type II laryngomalacia was diagnosed, together with localised tracheomalacia of the distal intrathoracic trachea. Urgent computed tomography (CT) with contrast was scheduled to exclude a mediastinal mass, vascular sling or ring. The scan revealed the presence of a curvilinear consolidation widening the oesophagus and displacing the trachea with significant narrowing at the level of the second to fifth thoracic vertebrae, consistent with an oesophageal foreign body (Fig 1). No other mediastinal or vascular anomalies were noticed. An urgent rigid oesophagoscopy was performed. During the procedure, food debris with thick mucus were removed piecemeal and suctioned (Fig 2). At 15cm, a pistachio shell was found and removed successfully with optical forceps (Fig 3). The child had an uneventful recovery. He was fed via a nasogastric tube for four days and was discharged once eating and drinking. The child was reviewed in clinic routinely and was found to be developing normally with resolution of the stridor and no upper airway signs or symptoms.

Figure 1.

A. Axial computed tomography (CT) image with contrast where the oesophageal foreign body could be seen as a hyperdense crescent-shaped lesion in the oesophagus. B. Coronal CT image.

Figure 2.

Oesophageal food bolus above the oesophageal foreign body.

Figure 3.

Foreign body – pistachio shell removed from the patient’s oesophagus.

Discussion

Foreign body ingestion is a common paediatric condition that is managed conservatively in the majority of cases. Occasionally, patients may require surgical intervention when symptoms are persistent and causing obstructive symptoms.² The problem generally occurs in children older than six months once motor skills have developed sufficiently, enabling objects to be placed in the mouth. Although patients frequently present with symptoms, in one case series of 325 children, 46% were asymptomatic at presentation and 54% presented with symptoms of dysphagia and pharyngeal or retrosternal pain.³ Other common presenting symptoms include refusal to eat and drooling. Respiratory symptoms are less common, but are usually associated with longstanding oesophageal foreign bodies and can lead to wheezing, stridor or cough.² Weight loss due to reduced calorific intake or recurrent aspiration pneumonia has also been reported. Sharp objects can perforate the oesophagus resulting in neck swelling, crepitus or pneumomediastinum.⁴ It is advised to retain a high index of suspicion for oesophageal foreign bodies when dealing with children with acute or chronic airway symptoms, especially in atypical presentations or when initial investigations findings fail to account for symptoms.

Our case is a presentation of chronic oesophageal foreign body where the absence of dysphagia, refusal to eat or drooling obscured the diagnosis. Additionally, our clinical impression was misdirected by the patient’s young age, as well as the lack of history of foreign body ingestion. In very young children who have yet to develop fine motor skills, a foreign body should always alert clinicians to the possibility of non-accidental injury. Suspicion should be further raised where there is inconsistency in the history, method of injury or a delayed presentation. With our case, an independent opinion was sought from a clinician with paediatric safeguarding experience, in keeping with local safeguarding guidelines. It was not thought that any foul play was involved, with the parents providing a credible history and having previously attempted to seek medical attention for their son. It appears that an older sister may have inadvertently placed the foreign body in the patient’s mouth, with the parents admitting that she occasionally tried to feed her younger brother. Nonetheless, it is important to educate parents to avoid similar events occurring again and that a record be kept for future reference with involvement of community services for continuing support.

Diagnostic microlaryngoscopy and bronchoscopy remain the gold standard investigations of choice to allow direct visualisation of the upper airway and allow for simultaneous intervention. The finding of localised narrowing of the trachea consistent with extraluminal compression is most commonly secondary to a vascular ring or sling. Vascular rings are caused by congenital abnormalities of the aortic arch and its major branches while vascular slings are due to an aberrant left pulmonary artery, causing deviation and compression of the distal trachea.⁵ The symptomatology of both conditions varies from airway obstruction and dysphagia to asymptomatic, found incidentally at a later age.

Where extraluminal compression is suspected on microlaryngoscopy and bronchoscopy, imaging is warranted to investigate the cause further. The choice of imaging should be made on a case by case basis and depends on local resources and practice. Both CT with angiography (CTA) and magnetic resonance imaging (MRI) may be used to evaluate the airway. While MRI does not carry the associated risks of radiation exposure, the prolonged examination time typically over 30 minutes necessitates general anaesthesia, preferably with a laryngeal mask airway, as endotracheal intubation could splint the trachea, masking any collapse or compression. This carries an inherent risk and is made more challenging when performed in a child with a potentially compromised airway. An algorithm has previously been described for deciding which patients would benefit from cross-sectional imaging.⁵ Our patient was able to tolerate CTA with sedation and this ruled out a vascular abnormality and revealed a probable oesophageal abnormality.

The management of stridor in the paediatric age group can be challenging. The first priority is always securing an airway, followed by a thorough history and examination including flexible laryngoscopy. While the majority of patients are managed conservatively, persistent or recurrent symptoms, decompensation and failure to thrive necessitate further investigation and management. The gold standard remains to be microlaryngoscopy and bronchoscopy with imaging in cases where extraluminal airway compression is suspected. Further management is targeted at the underlying cause.