Laryngopyocele in a case of bilateral mixed laryngocele: an impending airway emergency

Abstract

Laryngopyocele is a rare complication involving the laryngocele which can present with acute airway compromise. A 31-year-old man presented with acute onset respiratory distress and dysphagia. He had swelling on either side of upper aspect of the neck with tenderness on left side. Videolaryngoscopy using 70° rigid Hopkins rod telescope showed a swelling in the left pyriform sinus pushing the ipsilateral vocal cord. However, glottic space was adequate. Contract-enhanced CT scan of the neck confirmed left-sided mixed laryngopyocele with contralateral mixed laryngocele. Patient underwent excision of both the lesions in a single stage by transcervical approach. Laryngopyocele in a case of bilateral mixed laryngocele presenting as an impending airway emergency has not been reported in literature. The diagnostic and therapeutic challenges are discussed here along with review of literature.

Background

Laryngoceles are abnormal air-filled dilatation of laryngeal saccule communicating with the larynx.¹ Laryngomucoceles are formed when there is an obstruction in the neck of sac and the mucous secretion in saccule is retained. Infected laryngomucoceles are called laryngopyoceles and can present with acute airway emergency and/or dysphagia, with other symptoms being hoarseness, painful swelling in the neck and odynophagia. Incidence of laryngopyoceles in laryngoceles is around 5%–8%.² However, incidence of laryngopyoceles in bilateral laryngocele is not reported in literatures. In this report, authors have described a case of laryngopyocele in bilateral laryngocele which presented as impending airway obstruction. In view of the rarity of laryngopyoceles, and even more rare presentation of this lesion in a case of bilateral laryngocele with airway obstruction, authors have chosen to report this case. Presentation and management of this condition are discussed in this article, along with review of literature.

Case presentation

A 31-year-old man presented with respiratory difficulty and dysphagia for 3 days. The respiratory difficulty was sudden in onset, associated with intermittent noisy breathing, aggravated while lying in supine position and partially relieved in lateral decubitus position. Dysphagia was also sudden in onset, more for solids and associated with odynophagia and halitosis. He had history of swelling on either side of upper aspect of the neck for 2 months. Being an engineer with no other comorbid illness, he had no history of aspiration, regurgitation or hoarseness. On physical examination, he was tachycardic (pulse rate 110/min) and febrile (temperature 38.3°C). Respiratory rate was 19/min with no distress at rest. On examination of the neck, a 4×3 cm firm, tender, globular swelling was detected on the left side. The swelling was also warm and increased in size on Valsalva manoeuvre (figure 1). Another swelling of 2×3 cm was present on right side of the neck which was soft, non-tender and reducible. This swelling also increased in size on Valsalva and skin over the swelling was normal. On auscultation of chest, he had normal vesicular breath sounds with no other added sounds.

Figure 1.

Clinical picture of the patient showing a swelling on the left side of neck (red arrow).

Investigations

Videolaryngoscopy using a 70° rigid Hopkins rod telescope showed a smooth-surfaced swelling in the left pyriform sinus pushing the left true and false vocal cords. Both the vocal cords were mobile with adequate glottic space.

Contrast-enhanced CT (CECT) of the neck showed a 31×20×17 mm hypodense lesion with peripheral enhancement in the region of left pyriform fossa, suggestive of an abscess. It had both intralaryngeal and extralaryngeal components. Another lesion of size 20×10×5 mm was also seen on right side of the neck, which was well-defined and hypodense, similar to air, extending from the ventricles through the thyrohyoid membrane into the neck (figure 2). CECT chest was normal. White cell count was 2.18×10⁹/L with 78% neutrophil predominance. Erythrocyte sedimentation rate was 30 mm/hour.

Figure 2.

Coronal section of contrast-enhanced CT scan of neck on the left side. A well-defined air-filled smooth-walled sac could be seen, extending from paraglottic space through the thyrohyoid membrane into extralaryngeal region (red arrow). On the right side, a fluid-filled sac could be seen with similar extensions as the left side (green arrow).

Differential diagnosis

Swelling with increase in size on Valsalva manoeuvre, intralaryngeal and extra- laryngeal components in CT and videolaryngoscopy were suggestive of bilateral mixed laryngocele. The acute clinical presentations including neutrophilia and central hypodensity with peripheral rim enhancement in CT were suggestive of an underlying abscess. Based on these findings, authors diagnosed it as left-sided laryngopyocele with right-sided laryngocele.

However, infected saccular cyst, metastatic squamous cell carcinoma, branchial cleft cysts, lymphadenitis and cystic schwannomas could not be ruled out completely.

Treatment

Considering the risk of acute airway obstruction and aspiration, excision via external approach was planned. Written consent was obtained from the patient. Patient underwent excision of left laryngopyocele and right laryngocele by transcervical approach. The anaesthetist team successfully inserted endotracheal tube under fibre-optic laryngoscopic guidance without damaging the internal component of sac. A horizontal incision was given at the level of thyroid notch. After dissecting the subcutaneous tissues, sac of pyocele was opened and pus was drained. Then the cut-end of sac was held and dissected, tracking the sac through the thyrohyoid membrane till the level of saccule. Same procedure was repeated on the right side except for opening of sac (figure 3). The specimen was sent for histopathological examination.

Figure 3.

Intraoperative picture showing excision of the laryngoceles: left laryngopyocele (A) and right laryngocele (B).

Outcome and follow-up

Gastrograffin study to evaluate swallowing was done on third postoperative day. Subsequently, the patient was started on liquid diet followed by diet of semisolids and solids.

Histopathological examination of the specimen showed swellings lined by pseudostratified ciliated columnar epithelium with dense infiltration of neutrophils on the left side (figure 4).

Figure 4.

Histopathological examination (HPE) picture showing the pseudostratified ciliated columnar mucosal lining of the laryngocele (magnification 40×).

The patient is on regular follow-up for the last 6 months with no recurrence. He has been advised review after 6 months.

Discussion

Laryngopyoceles are rare complications of the laryngoceles, where retained mucus secretions are infected. They can present with dysphagia, hoarseness, neck swellings, septicaemia and respiratory distress. There are a few case reports in literature describing unilateral laryngopyocele as a cause of acute airway obstruction.^{3 4} However, a bilateral laryngocele with an underlying laryngopyocele as the cause of impending airway compromise is a rare coincidence and not reported so far.

Maweni et al have reported a similar case of laryngopyocele with contralateral laryngocele causing respiratory distress.⁵ The patient in their study presented with stridor and type 1 respiratory failure, requiring a combined approach for drainage of abscess. Although the patient in the reported study did not have an imminent respiratory failure, he had impending airway compromise and signs of sepsis which required emergency transcervical excision of the laryngopyocele.

Laryngoceles present a diagnostic dilemma with definitive diagnosis possible usually after radiology and endoscopy. Mallik et al reported laryngopyocele in an adult woman which mimicked an infected branchial cyst.⁶ The other lesions that mimic laryngopyoceles are infected saccular cyst, cervical lymphadenitis and extralaryngeal spread of aerodigestive malignancies.

Prasad et al and Lam and Lau have also reported cases of unilateral laryngopyocele causing airway obstruction.^{3 7} All these authors have used transcervical approaches to drain the abscess in view of the risk of aspiration.

Al-Yahya et al conducted a meta-analysis in 2016 and concluded that majority of surgeons preferred external approach for mixed laryngopyocele and endoscopic approach for internal pyocele. Out of three mixed laryngopyoceles operated endoscopically, one recurred and ultimately the surgeon opted for external approach during revision.⁸ The advantages of external approach are better visualisation and working space, along with minimal risk of aspiration. However, the merits have to be weighed against the demerits which include increased morbidity and prolonged hospital stay.

Laryngopyoceles can be life-threatening and four cases of mortality have been reported in literature.⁹ The probable causes for death in laryngopyocele are laryngeal inlet obstruction because of mass effect of the pyocele itself or discharge of pus into the larynx causing aspiration.

Töro et al had reported a fatal case caused due to asphyxia in a patient with laryngomucocele.¹⁰

Considering these possible fatalities, external approach is considered safe and effective for patients who present with acute or impending airway compromise. With the increased and widespread use of Laser, complete excision of laryngopyoceles is feasible with endoscopic approaches.¹¹ Use of suction diathermy and robots for laryngoceles is also reported in literature.¹²

The patient in this study did not have hoarseness which was unusual for the disease. He had neglected swellings in upper aspect of the neck and presented with acute respiratory distress. Treatment options for laryngocele are decided not only by size and extent but also by the way of presentation. In an acute emergency scenario with risk of fatal airway compromise, external approach has been considered safe. We managed to excise the lesion without tracheostomy in this case. Tracheostomy may be required in 10%–20% of laryngopyocele patients with acute airway compromise.⁸ In patients without emergency, endoscopic approach has been increasingly preferred even in cases of mixed laryngoceles, with Laser and Robotics making it resectable.

Patient’s perspective.

I am happy at the final outcome of treatment. I was having severe respiratory distress and then the doctors examined and told me that I may have a rare air-filled tumour in the larynx. The CT scan confirmed the problem. I was completely relieved of the symptoms within a week and I am happy to share my findings which might be useful in medical sciences and improving the patient care.

Learning points.

• Laryngopyocele in underlying bilateral mixed laryngocele is a rare cause of airway obstruction.

• CT and laryngoscopy are essential for definitive diagnosis and planning of treatment.

• Considering the risk of aspiration and airway obstruction, external approaches are considered safe and effective in management of mixed laryngopyocele.