Ann Thorac Surg 2004;78:1449-1451
© 2004 The Society of Thoracic Surgeons
Case report
Management of Bronchovascular Mucormycosis in a Diabetic: A Surgical Success
Vincent J. Reid, MDa,
Deborah L. Solnika,
Theodoris Daskalakis, MDa,
Kedambady P. Sheka, MDa,*
a Department of Surgery, Coney Island Hospital, Brooklyn, New York, USA
Accepted for publication July 3, 2003.
* Address reprint requests to Dr Sheka, Department of Surgery, Coney Island Hospital, 2601 Ocean Pkwy, Brooklyn, NY 11235, USA
shekak{at}nychhc.org
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Abstract
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Although cases of pulmonary mucor are scarce, diabetics account for a large percentage of these patients. The synergism of diabetes mellitus and mucormycosis poses potentially devastating bronchopulmonary complications, warranting urgent intervention. This report reviews the efficient workup, along with successful medical and surgical management, of a patient with pulmonary mucormycosis, with evidence of superior vena cava invasion.
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Introduction
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Mucor will almost never invade a person with adequate host defenses, but can cause fulminant disease in an individual with underlying conditions, including diabetes mellitus, organ transplantation, hematological malignancies, and chronic renal failure [1, 2]. Diabetics, especially with ketoacidosis, are specifically predisposed to this infection, not only because of the chemotactic defects of their polymorphic neutrophils, but also because mucor grows best in an acidic, hyperglycemic medium [3]. When the fungi colonize the lung, the hyphae may invade the pulmonary vasculature, resulting in severe ischemia, hemorrhage, and necrosis [3]. Once the diagnosis of pulmonary mucormycosis is made, a combination of medical and surgical therapy must be instituted without delay, in order to prevent critical complications, including fungal sepsis, respiratory failure, and massive pulmonary hemorrhage [2].
A 51-year-old male Mexican immigrant, with a 4-year history of uncontrolled diabetes and a 40-pack-year history of tobacco use, presented to the hospital complaining of 3 days of a dry, nonproductive cough, mild dyspnea, and malaise. There were no significant findings on physical examination. Serum and urine laboratory investigations revealed hyperglycemia and ketosis. The initial chest roentgenogram demonstrated a 4-cm density in the right upper lobe. A computed tomography scan of the chest localized the lesion to the medial segment of the right upper lobe, and showed evidence of extension towards the lateral wall of the superior vena cava (Fig 1).
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Fig 1. A computed tomography scan of the chest, at admission, displaying a mass in the right upper lobe, appearing to extend towards the lateral wall of the superior vena cava.
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The patient was treated appropriately for his diabetic ketosis, and the pulmonary lesion was further investigated. A fiberoptic bronchoscopy was performed, and a cheesy necrotic mass was seen in the medial right upper lobe. On microscopy, a tissue biopsy revealed nonseptate right-angle branching hyphae, typical of mucormycosis (Fig 2).
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Fig 2. The tissue biopsy, with a Giemsa stain, observed under medium power (x100) microscopy, demonstrating hyphae, characteristic of the mucor species.
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The patient was started on 1 mg/kg/d of Amphoteracin B. After 7 days of antifungal therapy, a repeated computed tomography scan demonstrated a cavitating lesion, with no evidence of caval infiltration (Fig 3). The patient was then scheduled for surgery. Through a standard posterolateral right thoracotomy incision, the right upper lobe was explored, and a cavitary lesion was discovered, confined to the medial segment (Fig 4). The superior vena cava was carefully dissected off of the upper lobe without much difficulty, confirming the disappearance of the caval invasion. The dissection was continued, and a right upper lobectomy was completed.
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Fig 3. A computed tomography scan, after 1 week of antifungal therapy, demonstrating some resolution of the right upper lobe mass.
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Postoperatively, Amphoteracin B was continued for 2 weeks. The patient remained stable, with no clinical or radiologic evidence of recurrence, and he was discharged on postoperative day 19. It has now been more than 6 months since the surgery, and the patient continues to be seen regularly in the thoracic surgery clinic. The patient remains disease-free, and has been more compliant with his diabetic medications.
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Comment
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An early diagnosis of pulmonary mucormycosis is difficult, because of the rarity of the disease, the variability of clinical presentation, the vagueness of radiologic findings, and a lack of sensitivity of cytologic assessment [4]. Given the patient's long history of smoking, a primary lung malignancy was suspected, but an infectious etiology was still investigated. The most common symptoms, signs, and radiologic presentations are no different than those of many major pulmonary diseases. As seen here, blood and sputum cultures are extremely insensitive. One study found that cultures from sputum samples or samples obtained from BAL, or from transthoracic or transbronchial needle aspiration, were diagnostic for only 1 out of 18 cases of pulmonary mucormycosis [5]. Therefore, the most widely used successful method of diagnosis is the visualization of the fungi upon microscopic examination, of diseased tissue collected during flexible fiberoptic bronchoscopy [2].
A recent, large retrospective study concluded that the in-hospital mortality for patients with isolated pulmonary mucormycosis was about 68%. Amphoteracin B is the drug of choice for mucormycosis, and should be started once the diagnosis is confirmed [2]. Recently, Amphoteracin B colloidal dispersion (ABCD) has shown to be an effective form of treatment in patients with renal failure [6].
The mortality of bronchovascular mucormycosis was reduced to 55% when medical treatment alone was provided, but patients treated surgically, with or without antifungal therapy, had a significantly lower mortality of approximately 27% [2]. There have been no large series performed to determine the appropriate time period between the initiation of medical therapy and surgical management. However, it is known that surgical therapy becomes less beneficial with increasing dissemination [2]. Consequently, if antifungal therapy appears to be effectual for bronchovascular mucormycosis, then a surgical plan should be implemented when there is evidence of regression of the invasion. The efficacy of computed tomography in the detection of fungal angioinvasion has been frequently stated in the literature. For this reason, computed tomography scans have altered the management of a number of cases [5].
In some cases, a wedge resection may suffice, but usually a lobectomy is necessary, and with more invasive disease, a pneumonectomy may be required [3]. Taking into consideration the presentation described in this case, and also the results of previously performed studies, the decision to treat this patient with a lobectomy was made, and was followed, without complications.
Pulmonary mucormycosis is a rare, but grave disease, with a predilection for uncontrolled diabetics. It is an acute and rapidly developing infection, which often results in fatality, if therapy is not provided promptly. This case report reviewed the successful medical and surgical intervention of a diabetic patient with an angioinvasive pulmonary mucor infection.
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References
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- Klemptner A. Pulmonary mucormycosis in a patient with COPD. Am Family Phys. 1999;59:2428[Medline]
- Adal KA, Lee FYW, Mussal SB. Pulmonary mucormycosis: the last 30 years. Arch Intern Med. 1999;159:183–185
- Anstadt MP, Hedge SS, Lowe JE, Spratt JA, Tedder M, Tedder SD. Pulmonary mucormycosis: results of medical and surgical treatment. Ann Thorac Surg. 1994;57:1044–1050[Abstract]
- Doo Yun L, Yoon Joo H, Young Sik P. Pulmonary mucormycosis. Asian Cardiovasc Thorac Ann. 2001;9:146–149[Abstract/Free Full Text]
- McAdams HP, Patz EF, Rosado de Christenson M, Strollo DC. Pulmonary mucormycosis: radiologic findings in 32 cases. Am J Radiol. 1997;168:1541–1548[Abstract/Free Full Text]
- Anaissie EJ, Andres E, Bowden RA, et al. Treatment of 21 cases of invasive mucormycosis with Amphoteracin B colloidal dispersion. Eur J Clin Infect Dis. 2001;20:460–466
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B. Spellberg, J. Edwards Jr., and A. Ibrahim
Novel Perspectives on Mucormycosis: Pathophysiology, Presentation, and Management
Clin. Microbiol. Rev.,
July 1, 2005;
18(3):
556 - 569.
[Abstract]
[Full Text]
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Abstract
Introduction
