INTRODUCTION: Mucormycosis is a fungal disease with high mortality and especially a rapid progression in pulmonary infections. Due to the high mortality, early and appropriate treatment is critical in the therapeutic success of patients with mucormycosis. Limited data are available regarding the ideal treatment for patients with mucormycosis. The following case illustrates a mucormycosis infection in a poorly controlled diabetic with a non ketoacidotic state treated with surgical resection and a combination of liposomal amphotericin B and echinocandin, which has not been previously reported for pulmonary mucormycosis.
CASE PRESENTATION: A 29 year old male with a medical history of smoking and type I diabetes mellitus of 10 years duration, was seen in the emergency department with an 8-week history of progressive shortness of breath, left sided pleuritic chest pain and progressive hemoptysis. He denied any fever, chills or night sweats. No intravenous drugs use or close contacts with persons with tuberculosis was reported. On exam he was afebrile with normal blood pressure, heart rate and respiratory rate. Oxygen saturation was 99% on room air. He had egophony and decreased breath sounds in the middle and lower left lung fields. Abdomen was soft, with the presence of a colostomy in the left lower quadrant. Laboratory data: normal complete blood cell count and urinalysis. Chemistry unremarkable, except for glucose 587 mg/dl and carbon dioxide 34mmol/L. Serum ketones negative. Hemoglobin A1C 10.2% and serum pH of 7.46. Coccidiomycosis, histoplasmosis, aspergillosis and crytococcus serologies were negative. Sputum acid fast bacillae, anti-neutrophil antibody and HIV were negative. Clinical course: Chest radiograph and computerized tomography (CT) revealed left upper lobe (LUL) cavitation and left lower lobe (LLL) consolidation. Bronchoscopy did not showed endobronchial lesions but LUL bronchial alveolar lavage (BAL) and transbronchial biopsies revealed non septated hyphae consistent with Rhizopus spp. CT scan of the head and sinus were normal and the patient was started on liposomal amphotericin B and micafungin intravenously on day 2. On day 4 he underwent LUL lobectomy plus LLL wedge resection with cultures from LUL positive for Rhizopus spp. There were no hyphae growth from the LLL resection and he completed 19 days of amphotericin B and micafungin. On day 20, he developed acute kidney injury, elevation of liver enzymes and pericardial tamponade. Antifungal therapy was stopped and a pericardial window was performed. There was no evidence of hyphae on the pericardial fluid. He recovered well and was started on posaconazole alone on day 25 to complete 4 additional weeks of oral antifungal therapy as outpatient.
DISCUSSION: Mucormycosis is a rare infection usually in patients with underlying immunocompromise. Diabetes mellitus especially with ketoacidosis is considered the most common underlying condition that favors this infection; however it is a less common association in pulmonary mucormycosis. The clinical presentation of fever and hemoptysis plus tissue infarction and necrosis are characteristic of pulmonary mucormycosis. Radiographic findings include focal consolidation, pleural effusions and multiple nodules, but cavitary lesions with an air crescent sign are rare. The standard therapy of pulmonary mucomycosis is the combination of surgical debridement and early antifungal therapy. Among antifungal, amphotericin B monotherapy has been the drug of choice before transitioning to oral agents. Rizhopus spp the most common form found in humans express the target enzyme for echinocandins. Therefore, combination of amphotericin B with echinocandins has been suggested from animals studies and a recent retrospective study performed in patients with rhino-orbital mucormycosis. However, no human data or case reports are available about the role of this antifungal combination in pulmonary mucormycosis.
CONCLUSIONS: Since the mortality of pulmonary mucormycosis is high, this case illustrates a successful outcome with an early aggressive combination of dual antifungal therapy and surgery.
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DISCLOSURE: The following authors have nothing to disclose: Juan Fernandez, Tamara Simpson, Marcos Restrepo
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