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Ann Thorac Surg 1997;63:230-232
© 1997 The Society of Thoracic Surgeons

Case Report

Tracheal Mucormycosis

David R. Andrews, MBBS, Andrew Allan, FRCS, Robert I. Larbalestier, FRACS

Department of Cardiothoracic Surgery, Royal Perth Hospital, Perth, Western Australia, Australia

Accepted for publication July 8, 1996.


   Abstract
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 Abstract
Introduction
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Pulmonary mucormycosis is a recognized entity occurring in diabetics and immunocompromised patients. It has a poor prognosis unless early diagnosis is made and appropriate surgical therapy instituted along with appropriate antifungal therapy. We describe here one of few cases of tracheal involvement by mucormycosis. Extensive destruction of the trachea and bibasal pneumonia led to the patient's death.


   Introduction
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Mucormycosis is a well-recognized fungal infection described in several forms [1]. It typically infects immunocompromised or diabetic patients [2]. The pulmonary form is characterized as an infection of the pulmonary parenchyma and larger bronchi typified by extensive vascular thrombosis and tissue necrosis. Few cases of upper tracheal/laryngeal mucormycosis have previously been reported [3].

A 75-year-old man presented to the emergency department unconscious, in severe diabetic ketoacidosis with additional systemic hypothermia and a history of dysphagia and progressive hoarseness.

Chest roentgenography demonstrated a right lower lobe consolidation. Sputum culture was negative for bacteria; no fungal elements were seen. His diabetic state was stabilized with acceptable but not ideal control of his blood sugar level between 7 and 15 mmol/L. Over the subsequent 3 weeks further investigations were performed. Video fluoroscopy demonstrated a lack of coordination of the oropharynx, with bilateral weakness and poor apposition of the vocal chords. Computed tomographic scan of his chest demonstrated a soft tissue mass encircling the trachea from the thoracic inlet to the tracheal bifurcation (Fig 1Go). Needle aspiration biopsy revealed reactive fibrous tissue.



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  		Fig 1. . Computed tomographic scan of the mediastinum showing the extensive circumferential tracheal mass.

 
At mediastinoscopy a soft, greyish-white mass was identified, with the consistency of chewing gum. Frozen section showed fungal elements with no malignancy. Further dissection was performed to obtain a larger representative specimen. During dissection the necrotic anterior wall of the trachea was breached, and in view of the extensive tracheal abnormality, the procedure was converted to a formal tracheostomy.

Histopathologic examination revealed necrotic debris surrounded by dense fibrosis with giant and small round cell infiltration. Broad (8- to 16-µm) frequently branching and nonseptate hyphae characteristic of mucormycosis were seen along with evidence of vessel thrombosis.

Subsequently copious volumes of sputum containing Pseudomonas aeruginosa and Enterobacter cloacae were aspirated from and around the tracheostomy. Intravenous administration of imipenem, tobramycin, and amphotericin B was commenced. Clinically the patient continued to deteriorate, and repeat computed tomographic scan demonstrated complete sloughing of the anterior trachea and infiltration of the great vessels (Fig 2Go).



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  		Fig 2. . Computed tomographic scan of the mediastinum showing the tracheal destruction. Note the migration of the tracheostomy tube.

 
The patient died of bacterial pneumonia. At autopsy a 55-mm length of upper trachea was found to be necrotic, the tracheal epithelium and cartilaginous rings having sloughed into the lumen of the trachea. This was surrounded by a space containing purulent material (Fig 3Go). Mucormycosis and bacteria were seen in and cultured from the autopsy specimen.



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  		Fig 3. . Postmortem photograph showing the extensive tissue destruction of the mid/lower trachea. Note the remnants of tracheal rings; just below these is the tracheal bifurcation.

 

   Comment
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 Introduction
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References
 
Pulmonary mucormycosis is associated with diabetes in approximately 32% of cases [1]. Virtually all other cases occur in patients with immunodeficiency or underlying malignancy. The fungus thrives in an acidotic, glucose-rich environment. In pulmonary mucormycosis the spores are inhaled, resulting in colonization with spread through the bronchial wall to invade blood vessels, especially arterioles, producing degrees of frank ischemia, hemorrhagic infarction, and tissue necrosis [1, 2].

Treatment has been classically delayed because of difficulty in establishing the diagnosis. Once the diagnosis is determined, amphotericin B is the most widely accepted medical therapy. Unless immediate clinical improvement is obtained, urgent and radical surgical intervention is indicated. A recent literature review on pulmonary mucormycosis revealed a mortality of 68% for medical therapy verses 11% for combined surgical and medical therapy [1].

Few previous cases of tracheal destruction by mucormycosis have been reported, although there are cases of mediastinal mucormycosis without tracheal involvement [4, 5]. Several case reports also document bronchial involvement by mucormycosis, either isolated or as a part of the pulmonary disease [6–8]. One previous case of mucormycosis involving the larynx and proximal trachea with minimal tissue destruction was successfully treated with a combination of operation and amphotericin B. The course of mediastinal mucormycosis appears to be similar to that of pulmonary mucormycosis, although it is harder to treat surgically, being not as easily debrided by removal of all or part of an organ. The tracheal destruction in our case was similar to but more extensive than that in previous reports involving the major bronchi [7, 8]. This may be explained by the added bacterial infection. Antifungal chemotherapy alone is clearly inadequate for the treatment of localized disease in the mediastinum, and a similar approach to that used in the lung is likely to yield better results, as evidenced by the previous successful case report [3].

Prompt and accurate diagnosis is vital to limit the extent of tissue destruction before operation; therefore, early surgical biopsy of atypical mediastinal and pulmonary masses should be sought, especially in diabetic and immunocompromised patients. This allows surgical intervention before the local tissue destruction is extensive and the patient's clinical condition deteriorates. The extent of tracheal destruction in our patient precluded resection of the disease, and his physical condition was prohibitive of major tracheal reconstruction.


   Footnotes
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Address correspondence to Mr Andrews, 48 Burnell St, Russell Lea NSW 2046, Australia.


   References
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 Abstract
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 References
 

  1. Tedder M, Spratt JA, Anastadt MP, et al. Pulmonary mucormycosis: results of medical and surgical therapy. Ann Thorac Surg 1994;57:1044–50.[Abstract]
  2. Sugar AM. Agents of mucormycosis and related species. In: Mandell GL, Bennett JE, Dolin R, eds. Mandell, Douglas and Bennett principles and practice of infectious diseases, 4th ed. New York: Churchill Livingston, 1995;2311--21.
  3. Schwartz JRL, Nagle MG, Elkins RC, Mohr JA. Mucormycosis of the trachea. Chest 1982;81:653–4.[Abstract/Free Full Text]
  4. Leong ASY. Granulomatous mediastinitis due to Rhizopus species. Am J Clin Pathol 1978;70:103–7.[Medline]
  5. Connor BA, Anderson RJ, Smith JW. Mucor mediastinitis. Chest 1979;75:524–6.[Abstract/Free Full Text]
  6. Dillon ML, Sealy WC, Fetter BF. Mucormycosis of the bronchus successfully treated by lobectomy. J Thorac Surg 1958;35:464–8.
  7. Winston RM. Phycomycosis of the bronchus. J Clin Pathol 1965;18:729–31.[Abstract/Free Full Text]
  8. Brown RM, Johnson JH, Kessinger JM, Sealy WC. Bronchovascular mucormycosis in the diabetic: an urgent surgical problem. Ann Thorac Surg 1992;53:854–5.[Abstract]



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This Article
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Robert I. Larbalestier
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Right arrow Articles by Larbalestier, R. I.

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