Case Reports

Infected Bronchogenic Cyst With Left Atrial, Pulmonary Artery, and Esophageal Compression

Fed Pract. 2020 April;37(4):190-193

References

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Discussion

Bronchogenic cysts can present at birth or later in life; patients may be asymptomatic for decades prior to discovery.⁴Cysts located in the mediastinum can cause compression of the trachea and esophagus and cause cough, dyspnea, chest pain, and dysphagia.⁵ More life-threatening complications include infection, tracheal compression, malignant transformation, superior vena cava syndrome, or spontaneous rupture into the airway.^6,7

Infection can occasionally occur, and various bacterial etiologies have been described. Hernandez-Solis and colleagues describe 12 cases of superinfected bronchogenic cysts with Staphylococcus aureus and Pseudomonas aeroginosa, the most commonly described organisms.⁸ Casal and colleagues describe a case of α-hemolytic Streptococci treated with amoxicillin.⁹ Liman and colleagues describe 2 cases of bronchogenic cyst infected with Mycobacterium and cite an additional case report by Lin and colleagues similarly infected by Mycobacterium.^10,11 Only 1 case was identified to have direct bronchial communication as a potential source of introduction of infection into bronchogenic cyst. In other cases, potential sources of infection were not identified, though it was postulated that direct ventilation could be a potential source of inoculation.

Surgical resection of mediastinal bronchogenic cysts has traditionally been considered the definitive treatment of choice.^12,13 However, bronchogenic cysts may sometimes be difficult to differentiate from soft tissue tumors by chest CT, especially in cases of cysts with nonserous fluid. In particular, cysts that are infected are likely to have increased density and high attenuation on imaging; therefore, surgical excision may be delayed until diagnosis is made.¹⁴ Due to low complication rates, EBUS is increasingly used in the diagnosis and therapeutic management of bronchogenic cysts as an alternative to surgery, particularly for those who are symptomatic.^15,16 Ultrasound guidance can allow for complete aspiration of the cyst, causing complete collapse of the cystic space and can facilitate adhesion between the mucosal surfaces lining the cavity and reduce recurrence.¹⁷ Nonetheless, bronchogenic cysts that are found to be infected, recur, or have a malignant component should be resected for definitive treatment.¹⁸

The mass discovered on our patient’s imaging appeared to have heterogenous attenuation consistent with malignancy rather than homogenous attenuation surrounded by a clearly demarcated wall consistent with a cystic structure; therefore, EBUS-TBNA was initially pursued and yielded an expedited diagnosis of the first-ever described bronchogenic cyst with Actinomyces superinfection as well as dramatic symptomatic relief of compression of surrounding mediastinal structures, particularly of the right pulmonary artery. As this is a congenital malformation, the patient was likely asymptomatic until the cyst became infected, after which he likely experience cyst growth with subsequent encroachment of surrounding mediastinal structures. Additionally, identification of pathogen by TBNA allowed for treatment before surgical excision, possibly avoiding accidental spread of pathogen intraoperatively.

Conclusions

Our case adds to the literature on the use of EBUS-TBNA as a diagnostic and therapeutic modality for bronchogenic cyst. While cases of mediastinitis and pleural effusion following EBUS-guided aspiration of bronchogenic cysts have been reported, complications are extremely rare.¹⁹ EBUS is increasingly favored as a means of immediate diagnosis and treatment in cases where CT imaging may not overtly suggest cystic structure and in patients experiencing compression of critical mediastinal structures.