Tracheal perforation and subcutaneous emphysema secondary to pseudomembranous invasive Aspergillus tracheobronchitis successfully managed with endotracheal tube manipulation

Kazushige Shiraishi1, Takkin Lo2,3

1 Department of Medicine, John A. Burns School of Medicine, University of Hawaii, Honolulu, USA

2 Pulmonary Medicine, Adventist Health Castle Medical Center, Kailua, USA

3 Hawaii Pulmonary Health Center, Kailua, USA

Corresponding author’s e-mail: kshira@hawaii.edu

Received: 23/12/2024
Accepted: 30/12/2024
Published: 22/01/2025

Conflicts of Interests: The Authors declare that there are no competing interests.
Patient Consent: Consent was obtained from the patient’s family.
Acknowledgements: We thank the patient and family for taking part in the study. An earlier version of this work was presented as an oral presentation at the CHEST annual conference, 2024 (Shiraishi K, Lo T. An Unusual Case Of Subcutaneous Emphysema Secondary To Aspergillus Tracheobronchitis: What Now? 2024;166:A1046-A1047. Doi:10.1016/j.chest.2024.06.693).
This article is licensed under a Commons Attribution Non-Commercial 4.0 License

How to cite this article: Shiraishi K, Lo T. Tracheal perforation and subcutaneous emphysema secondary to pseudomembranous invasive Aspergillus tracheobronchitis successfully managed with endotracheal tube manipulation. EJCRIM 2025;12:doi:10.12890/2025_005118

ABSTRACT

Introduction: Aspergillus is a ubiquitous fungus causing various pulmonary diseases depending on the host’s immune status. Aspergillus tracheobronchitis, a rare form of invasive aspergillosis, primarily affects severely immunocompromised or critically ill patients. We present the first known case of Aspergillus tracheobronchitis complicated by tracheal perforation and subcutaneous emphysema successfully treated with conservative endotracheal tube manipulation.
Case description: A 64-year-old male with type 2 diabetes mellitus presented with generalized weakness and abdominal discomfort, later diagnosed with a perforated cecum requiring right colectomy. His postoperative course in the intensive care unit was complicated by septic shock, acute kidney injury, and failed extubation due to airway compromise. Seven days after the failed extubation, he developed subcutaneous emphysema in the neck. Chest computed tomography scan showed neck, left chest wall, and mediastinal emphysema. Bronchoscopy identified a focal black necrotic lesion on the left proximal tracheal wall and multiple small mucosal ulcerations throughout the proximal to distal trachea. The endotracheal tube was advanced beyond proximal tracheal necrotic lesion. Subcutaneous emphysema reduced overnight, suggesting that the lesion was the source of the air leak. Bronchial washings confirmed Aspergillus fumigatus, establishing a diagnosis of invasive Aspergillus tracheobronchitis. Treatment with voriconazole prevented further expansion of emphysema, which gradually resolved.
Conclusion: Subcutaneous emphysema in ventilated patients with tracheobronchitis is a rare and challenging complication. This case demonstrates successful management through endotracheal tube manipulation to tamponade the lesion, highlighting subcutaneous emphysema as a potential manifestation of Aspergillus tracheobronchitis and offering a minimally invasive treatment approach.

KEYWORDS

Aspergillus, invasive Aspergillosis, Aspergillus tracheobronchitis, subcutaneous emphysema, mediastinal emphysema

LEARNING POINTS

INTRODUCTION

Aspergillus is a ubiquitous fungus that can lead to a variety of pulmonary conditions depending on the host’s immune status. Common forms of aspergillosis include allergic bronchopulmonary aspergillosis, invasive aspergillosis, chronic necrotizing aspergillosis, and aspergilloma. Aspergillus tracheobronchitis, a rare form of invasive aspergillosis, typically occurs in critically ill or severely immunocompromised patients and only rarely causes tracheal perforation and subcutaneous emphysema[1,2]. Here, we present the first known case of Aspergillus tracheobronchitis complicated by subcutaneous emphysema of the neck and chest wall, along with mediastinal emphysema that was successfully managed using conservative manipulation of the endotracheal tube, without the need for surgical intervention.

CASE DESCRIPTION

A 64-year-old male non-smoker with a history of hypertension, type 2 diabetes mellitus, chronic kidney disease, deep venous thrombosis, and leg osteomyelitis presented with generalized weakness, dizziness, and abdominal discomfort for one week. An initial computed tomography (CT) scan of the abdomen and pelvis did not reveal any abnormalities to explain his abdominal symptoms. Ten days later, due to persistent abdominal discomfort, an abdominal and chest X-ray was performed, which identified pneumoperitoneum. The patient was urgently taken to the operating room and underwent a right colectomy for a perforated cecum. The patient’s postoperative course in the intensive care unit was complicated by septic shock requiring multiple vasopressors and systemic steroids, acute kidney injury necessitating continuous renal replacement therapy, and anemia with thrombocytopenia requiring massive transfusions of packed red blood cells, fresh frozen plasma, and platelets. Following surgery, the patient remained on mechanical ventilator for 6 days and was successfully extubated for about six hours before needing to be reintubated due to airway compromise. A plan was made to perform a tracheostomy for failed extubation and the inability to maintain airway. Seven days after the reintubation, the patient developed upper chest subcutaneous emphysema. Despite the absence of documented traumatic reintubation or oral bloody secretions, a tracheal perforation was suspected, prompting pulmonology consultation.

Physical examination revealed a well-nourished male intubated on the mechanical ventilator with fraction of inspired oxygen (FiO2) of 24%. His blood pressure was 133/51 mmHg, with a normal temperature and pulse. Neck examination revealed subcutaneous emphysema bilaterally. Cardiac and pulmonary examinations were unremarkable. The abdomen was soft and non-tender, with a Jackson-Pratt drain in place. Bilateral pitting edema was noted. Laboratory analysis (Table 1) was remarkable for hemoglobin level of 7.6 g/d, platelet count of 19 ×103/μl, total protein of 4.7 g/dl, total bilirubin of 12.9 mg/dl, and a prothrombin time-international normalized ratio (PT-INR) of 1.75. A chest X-ray demonstrated subcutaneous gas in the neck and left chest wall, along with previously identified airspace disease in the right lung (Fig. 1). Chest CT scan confirmed the presence of gas within the soft tissues of the neck, chest, and mediastinum (Fig. 2).

(click to enlarge)

Table 1. Laboratory data.

(click to enlarge)

Figure 1. Chest X-ray showed subcutaneous gas in the neck and left chest wall, along with the previously identified airspace disease in the right lung. Yellow arrows indicate subcutaneous gas.

(click to enlarge)

Figure 2. Chest CT showed gas in the neck, chest, and mediastinum. Lower zone (A) and upper zone (B) of the lungs were visualized. Yellow arrows indicate gas.

Bronchoscopy showed multiple punctate mucosal ulcerations, each measuring approximately 0.5 × 0.5 cm, with necrotic center and a whitish surrounding border, distributed throughout the proximal to distal trachea (Fig. 3A). Additionally, a focal area of black discoloration with necrosis was observed on the left lateral wall of the proximal trachea, just below the subglottic region. This lesion measured approximately 1 x 2 cm and was notable for whitish pseudomembranous mucoid center (Fig. 3B). This was presumed to be the cause of subcutaneous emphysema in the left chest. To tamponade the lesion and progression of emphysema, the endotracheal tube was advanced past the proximal tracheal lesion to rest approximately 3 cm above the carina. Subcutaneous emphysema reduced overnight, further suggesting that the lesion was the source of the emphysema. Fungal culture for bronchial washing was positive for A. fumigatus, establishing the diagnosis of Aspergillus tracheobronchitis and invasive pulmonary Aspergillosis. Treatment with voriconazole was initiated, and the subcutaneous emphysema remained stable and gradually resolved over time. Given the prolonged hospitalization and a guarded prognosis, the patient and family opted to transition to palliative care, and the patient was discharged to an inpatient hospice program.

(click to enlarge)

Figure 3. Bronchoscopy revealed multiple mucoid ulcers and a necrotic lesion in the proximal trachea. A) Multiple punctate mucosal ulcerations, each measuring approximately 0.5 × 0.5 cm, with necrotic centers and whitish borders, are scattered throughout the trachea from the proximal to distal segments. B) Black arrows indicate ulcerations. A necrotic lesion measuring up to 1 × 2 cm is noted on the left lateral wall of the proximal trachea, just below the subglottic region, with a pseudomembranous whitish mucoid center. This lesion was thought to be the cause of subcutaneous emphysema. Yellow arrow indicates necrotic area.

DISCUSSION

This is the first reported case of tracheal perforation and subcutaneous emphysema secondary to Aspergillus tracheobronchitis successfully managed with conservative manipulation of the endobronchial tube. We performed a systematic search on MEDLINE and Google Scholar, using the following keywords: “tracheal perforation,” “subcutaneous emphysema,” “Aspergillus tracheobronchitis,” and “endotracheal manipulation.” This search yielded two case reports describing tracheal perforation secondary to pseudomembranous Aspergillus tracheobronchitis[1,3] and two case reports of tracheal perforation involving unspecified (non-pseudomembranous) Aspergillus tracheobronchitis[4,5]. Notably, none of these publications documented treatment with endotracheal tube manipulation, and only one case report documented subcutaneous emphysema associated with Aspergillus tracheobronchitis[1]. Tracheobronchitis is a rare but serious manifestation of invasive Aspergillosis, with reported overall mortality rate of 48%[6]. Infection or traumatic disruption of the tracheal mucosa can lead to subcutaneous emphysema[7], and in our case, Aspergillus tracheobronchitis was identified as the underlying cause. A. fumigatus is the most common Aspergillus species that cause tracheobronchitis[8,9], and in our case A. fumigatus was detected. Aspergillus tracheobronchitis presents in three forms: ulcerative, pseudomembranous, and obstructive[10]. Our patient had multiple tracheal ulcers as well as a focal area of black necrotic lesion with pseudomembranous mucoid center, the latter was thought to be responsible for the development of subcutaneous emphysema; supported by the observation that the subcutaneous emphysema improved following endotracheal tube adjustment to tamponade the responsible lesion. Managing subcutaneous emphysema caused by a tracheal lesion in a ventilated patient is challenging. In our case, it was successfully managed with endotracheal tube advancing for 3 cm above the carina, effectively to tamponading the air leak, in combination with voriconazole.

Early diagnosis and treatment of Aspergillus tracheobronchitis are crucial due to its high mortality rate, particularly of pseudomembranous and obstructive forms, where mortality can reach up to 80%[11]. Nevertheless, diagnosis remains challenging as Aspergillus tracheobronchitis lacks specific clinical symptoms and distinctive radiological findings. Aspergillus tracheobronchitis is frequently associated with lung transplant[8,12], acquired immunodeficiency syndrome (AIDS)[13], diabetes mellitus[14,15], chronic obstructive pulmonary disease[15], and malignancies[9]. In our case, the patient had diabetes, was receiving systemic corticosteroids for severe sepsis, and required intensive care unit admission with mechanical ventilation; all factors predisposing to invasive Aspergillosis. Early utilization of bronchoscopy in patients with such risk factors is essential for the timely diagnosis of Aspergillus tracheobronchitis.

Despite the current recommendation to use antifungal agents for invasive Aspergillosis[16], no standardized protocol exists for managing tracheal wall perforation and subcutaneous emphysema associated with tracheobronchitis. Most studies on tracheal perforation primarily address the management of tracheobronchial injuries[17], rather than perforations caused by tracheobronchitis. Patients with severe tracheobronchitis frequently have immunocompromising risk factors, which further complicates treatment strategies. Potential management options include tracheostomy, tracheal reconstruction, or the use of tracheal stents, all of which necessitate multidisciplinary discussions. The development of a registry to collect and analyze similar cases is essential for establishing evidence-based treatment protocols.

References

  1. Khoo K-L, Eng P. Tracheobronchial Aspergillosis. J Bronchology Interv Pulmonol 2001;8:32-33.
  2. Kousha M, Tadi R, Soubani AO. Pulmonary aspergillosis: a clinical review. Eur Respir Rev 2011;20:156-74.
  3. Argüder E, Şentürk A, Hasanoğlu HC, Hasanoğlu İ, Kanbay A, Doğan HT. Unique Case of Pseudomembranous Aspergillus Tracheobronchitis: Tracheal Perforation and Horner’s Syndrome. Mycopathologia 2016;181:885-889.
  4. Aerni MR, Parambil JG, Allen MS, Utz JP. Nontraumatic disruption of the fibrocartilaginous trachea: causes and clinical outcomes. Chest 2006;130:1143-1149.
  5. Gonzalez M, Ma Tooh M, Krueger T, Ris HB, Letovanec I, Wang Y, et al. Repair of tracheal aspergillosis perforation causing tension pneumothorax. Ann Thorac Surg 2013;96:2256-2258.
  6. Karnak D, Avery RK, Gildea TR, Sahoo D, Mehta AC. Endobronchial fungal disease: an under-recognized entity. Respiration 2007;74:88-104.
  7. Maunder RJ, Pierson DJ, Hudson LD. Subcutaneous and mediastinal emphysema. Pathophysiology, diagnosis, and management. Arch Intern Med 1984;144:1447-1453.
  8. Mehrad B, Paciocco G, Martinez FJ, Ojo TC, Iannettoni MD, Lynch JP 3rd. Spectrum of Aspergillus infection in lung transplant recipients: case series and review of the literature. Chest 2001;119:169-175.
  9. Wu N, Huang Y, Li Q, Bai C, Huang HD, Yao XP. Isolated invasive Aspergillus tracheobronchitis: a clinical study of 19 cases. Clin Microbiol Infect 2010:16:689-695.
  10. Denning DW. Commentary: unusual manifestations of aspergillosis. Thorax 1995;50:812-813.
  11. Tasci S, Glasmacher A, Lentini S, Tschubel K, Ewig S, Molitor E, et al. Pseudomembranous and obstructive Aspergillus tracheobronchitis - optimal diagnostic strategy and outcome. Mycoses 2006;49:37-42.
  12. Kramer MR, Denning DW, Marshall SE, Ross DJ, Berry G, Lewiston NJ, et al. Ulcerative tracheobronchitis after lung transplantation. A new form of invasive aspergillosis. Am Rev Respir Dis 1991;144:552-556.
  13. Kemper CA, Hostetler JS, Follansbee SE, Ruane P, Covington D, Leong SS, et al. Ulcerative and plaque-like tracheobronchitis due to infection with Aspergillus in patients with AIDS. Clin Infect Dis 1993;17:344-152.
  14. Chang SM, Kuo HT, Lin FJ, Tzen CY, Sheu CY. Pseudomembranous tracheobronchitis caused by Aspergillus in immunocompromised patients. Scand J Infect Dis 2005;37:937-942.
  15. He H, Jiang S, Zhang L, Sun B, Li F, Zhan Q, et al. Aspergillus tracheobronchitis in critically ill patients with chronic obstructive pulmonary diseases. Mycoses 2014;57:473-482.
  16. Patterson TF, Thompson GR 3rd, Denning DW, Fishman JA, Hadley S, Herbrecht R, et al. Practice Guidelines for the Diagnosis and Management of Aspergillosis: 2016 Update by the Infectious Diseases Society of America. Clin Infect Dis 2016;63:e1-e60.
  17. Grewal HS, Dangayach NS, Ahmad U, Ghosh S, Gildea T, Mehta AC. Treatment of Tracheobronchial Injuries: A Contemporary Review. Chest 2019;155:595-604.