Isolated tracheobronchial mucormycosis: Report of a case and systematic review of literature

Mycoses. 2023 Jan;66(1):5-12. doi: 10.1111/myc.13519. Epub 2022 Sep 1.

Abstract

Background: Isolated tracheobronchial mucormycosis (ITBM) is an uncommonly reported entity. Herein, we report a case of ITBM following coronavirus disease 2019 (COVID-19) and perform a systematic review of the literature.

Case description and systematic review: A 45-year-old gentleman with poorly controlled diabetes mellitus presented with cough, streaky haemoptysis, and hoarseness of voice 2 weeks after mild COVID-19 illness. Computed tomography and flexible bronchoscopy suggested the presence of a tracheal mass, which was spontaneously expectorated. Histopathological examination of the mass confirmed invasive ITBM. The patient had complete clinical and radiological resolution with glycaemic control, posaconazole, and inhaled amphotericin B (8 weeks). Our systematic review of the literature identified 25 additional cases of isolated airway invasive mucormycosis. The median age of the 26 subjects (58.3% men) was 46 years. Diabetes mellitus (79.2%) was the most common risk factor. Uncommon conditions such as anastomosis site mucormycosis (in two lung transplant recipients), post-viral illness (post-COVID-19 [n = 3], and influenza [n = 1]), and post-intubation mucormycosis (n = 1) were noted in a few. Three patients died before treatment initiation. Systemic antifungals were used in most patients (commonly amphotericin B). Inhalation (5/26; 19.2%) or bronchoscopic instillation (1/26; 3.8%) of amphotericin B and surgery (6/26; 23.1%) were performed in some patients. The case-fatality rate was 50%, primarily attributed to massive haemoptysis.

Conclusion: Isolated tracheobronchial mucormycosis is a rare disease. Bronchoscopy helps in early diagnosis. Management with antifungals and control of risk factors is required since surgery may not be feasible.

Keywords: CAM; airway aspergillosis; bronchus; mucorales; pulmonary mucormycosis.

Publication types

  • Systematic Review
  • Case Reports
  • Review

MeSH terms

  • Amphotericin B / therapeutic use
  • Antifungal Agents / therapeutic use
  • COVID-19* / complications
  • Female
  • Hemoptysis / drug therapy
  • Humans
  • Male
  • Middle Aged
  • Mucormycosis* / diagnosis
  • Mucormycosis* / drug therapy

Substances

  • Amphotericin B
  • Antifungal Agents