Table of Contents  
CASE REPORT
Year : 2016  |  Volume : 9  |  Issue : 1  |  Page : 132-135  

Endobronchial pulmonary mucormycosis diagnosed by fiberoptic bronchoscope: A rare case report


1 Department of Pulmonary Medicine, KLE University's J.N. Medical College, Belgaum, Karnataka, India
2 Department of Womens Health, Institute of Physiotherapy, KLE University, Belgaum, Karnataka, India
3 Department of Pathology, KLE University's J.N. Medical College, Belgaum, Karnataka, India

Date of Web Publication22-Dec-2015

Correspondence Address:
Vinay Mahishale
Department of Pulmonary Medicine, KLE University's J.N. Medical College, Belgaum, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-2870.167991

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  Abstract 

Pulmonary mucormycosis is relatively uncommon, but a life-threatening infection affecting mostly individuals with diabetes mellitus, hematological malignancies, chronic renal failure, posttransplantation and other immunocompromised states. Mucormycosis of the lung has a wide range of clinical and radiological manifestations. Very few cases of pulmonary mucormycosis presenting as a fungal ball, cavity resembling tuberculosis, nonresolving, and recurrent pneumonia in patients with diabetes mellitus or other immunosuppressive conditions, were reported from India. We report a case of pulmonary mucormycosis in an adult male patient with uncontrolled diabetes.

Keywords: Bronchoscope, enobronchial, mucormycosis, pulmonary


How to cite this article:
Mahishale V, Patil B, Mahishale A, Malur PR, Avuthu S, Eti A, Lolly M. Endobronchial pulmonary mucormycosis diagnosed by fiberoptic bronchoscope: A rare case report . Med J DY Patil Univ 2016;9:132-5

How to cite this URL:
Mahishale V, Patil B, Mahishale A, Malur PR, Avuthu S, Eti A, Lolly M. Endobronchial pulmonary mucormycosis diagnosed by fiberoptic bronchoscope: A rare case report . Med J DY Patil Univ [serial online] 2016 [cited 2020 Sep 18];9:132-5. Available from: http://www.mjdrdypu.org/text.asp?2016/9/1/132/167991


  Introduction Top


Pulmonary mucormycosis is relatively uncommon, but a life-threatening infection affecting mostly individuals with diabetes mellitus, hematological malignancies, chronic renal failure, posttransplantation, and other immunocompromised states. The first case of pulmonary mucormycosis was reported by Furbinger in 1876. [1] Mucormycosis of the lung has a wide range of clinical and radiological manifestations. Few cases of pulmonary mucormycosis presenting as a fungal ball, cavity resembling tuberculosis (TB), nonresolving, and recurrent pneumonia in patients with diabetes mellitus or other immunosuppressive conditions, were reported from India. [2],[3] However, some cases are reported in immunocompetant individuals as well. [4]


  Case Report Top


A 44-year-old man was admitted third time with few days history of fever, shortness of breath, cough and hemoptysis. The patient's initial presentation was 3 months earlier. He was then diagnosed as a case of community-acquired pneumonia and was discharged on oral antibiotics. He was admitted again 3 weeks prior to this current admission for the same symptoms. He was diagnosed as health care-associated pneumonia and treated accordingly. During the last admission, TB and HIV infections were ruled out. His medical history was not significant except uncontrolled diabetes since 7 years. He had no history of smoking, alcohol consumption or IV drug abuse. On physical examination, the patient was a healthy looking man in mild respiratory distress. Vital signs were as follows: Temperature 38.8°C; pulse rate 96/min; blood pressure 110/82 mmHg; respiratory rate 24/min and oxygen saturation 94% at room air.

Patients laboratory findings included white blood cell (WBC) count 7900/dl, Hb 13.2 g/dl and platelet count 316,000. Sputum and blood cultures from all three admissions were negative including serologies for Q fever and Brucella. Renal functions, electrolytes, liver enzymes, and coagulation profiles were within normal limits. However, his fasting and postprandial blood sugars were 210 mg/dl and 362 mg/dl respectively. HbA1c was as high as 12%.

The chest radiograph revealed evidence of consolidation with air bronchograms in the left lower zone [Figure 1]. Previous two admissions had similar radiological features. Contrast enhancing computed tomography (CT) of thorax revealed 4.2 cm × 4 cm thick rim enhancing lesion in the left lower lobe with central cavitatory necrosis and small air fluid level suggestive of abscess [Figure 2]. Fiberoptic bronchoscopy revealed thickened edematous left lower lobe bronchus with significant narrowing. A valve-like neoplasm was seen from which biopsy was obtained. Histological sections showed plenty of fungal hyphae in a necrotic inflammatory background with the absence of granuloma or malignancy. Giemsa-stained smear showed broad, irregular, nonseptate hyphe with right-angled branching consistent with the diagnosis of mucormycosis [Figure 3]. Gomori's methenamine silver staining of the same demonstrated the typical hyphae suggestive of mucormycosis [Figure 4]. Fungal culture of the specimen inoculated on Saboraud's dextrose agar medium yielded white colonies within 3 days and organism was identified as mucor. As the patient was not willing for surgery, we started treatment with intravenous liposomal amphotericin B at a dose of 5 mg/kg for a period of 4 weeks. Fever subsided after 4 days of starting liposomal amphotericin B and follow-up chest X-ray and CT scan after 6 weeks showed good resolution.
Figure 1: The chest radiograph revealed evidence of consolidation with air bronchograms in the left lower zone

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Figure 2: Contrast enhancing computed tomography of thorax revealed 4.2 cm × 4 cm thick rim enhancing lesion in the left lower lobe with central cavitatory necrosis and small air fluid level suggestive of
abscess


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Figure 3: Histological sections showed plenty of fungal hyphae in a necrotic inflammatory background with absence of granuloma or malignancy. Giemsa stained smear showed broad, irregular, nonseptate hyphe with right-angled branching consistent with the diagnosis of mucormycosis

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Figure 4: Gomori's methenamine silver staining of the same demonstrated the typical hyphae suggestive of mucormycosis

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  Discussion Top


Mucormycosis, also called zygomycosis is an important opportunistic infection caused by a fungus that belongs to the class zygomycetes, which is the third most common invasive fungal infection after candidiasis and aspergillosis. Although the infection rate of mucormycosis is very low, patients with immunosuppression, diabetes mellitus or hematological malignancy are at the highest risk for mucormycosis. [5] Mucorales, which belong to the subclass zygomycetes of the class phycomycetes, are opportunistic fungi that cause deep tissue infection. The production of spores, which become airborne, leads to the primary route of inoculation in the respiratory tract. The spores of mucorales show minimal pathogenicity in immunocompetent hosts because the macrophages can kill spores by phagocytosis and oxidative killing mechanisms. However, macrophages in a host with immune deficiency or diabetes can lose their ability to inhibit spore germination and prevent the hyphae and spores from invading the bronchus and lung. [6]

Pulmonary mucormycosis has an acute onset and a wide range of clinical manifestations, such as fever (most common), cough, hemoptysis, and dyspnea. Hemoptysis was found in about 25-30% of the patients. Mucormycosis is usually seen in diabetic patients with or without ketoacidosis, or in patients with other risk factors, including cancer (such as leukemia, lymphoma, multiplemyeloma, etc.), AIDS, chemotherapy, organ transplantation, anti-rejection treatment, iron chelate treatment, severe malnutrition and the application of broad-spectrum antibiotics. Clinically observed forms of mucormycosis are rhinocerebral, pulmonary, cutaneous, gastrointestinal, disseminated and other clinical manifestations. The most commonly involved mucormycosis is rhinocerebral and pulmonary forms. [7]

The clinical manifestations of pulmonary mucormycosis infection cannot be easily distinguished from those of pulmonary bacterial infection. Patients may present with fever, cough, expectoration, hemoptysis, and pleuritic chest pain. The laboratory examination results are nonspecific, with most patients only showing an increase in their WBC count. The radiographic presentations of patients with pulmonary mucormycosis appear abnormal and include infiltrate, cavity, consolidation, air crescent sign, pleural effusions, fistula, pneumothorax and pulmonary collapse. [8]

In few patients, the disease manifests as airway obstruction as the fungus invades bronchus and forms intrabronchial lesions even though chest radiography shows mediastinal widening or pulmonary atelectasis. Bronchoscopy appears to be a successful diagnostic modality in diagnosing mucormycosis with major airway involvement. Bronchoscopy reveals granulation tissue and grey-white mucoid material that frequently blocks a major airway. The airways involved are typically edematous and necrotic, or lesions with an appearance suggestive of a bronchial adenoma. In many cases, it is postulated that a submucosal, invasive fungal infection caused a submucosal abscess, which presented as an endobronchial mass. [9],[10] Reverse halo sign is a diagnostic radiological clue toward a diagnosis of mucormycosis.

As mucormycosis is an angioinvasive organism, it has a tendency to grow toward blood vessels, and a mass or nodule encroaching toward the great vessels of the mediastinum in successive radiographs suggests the possibility of mucormycosis. The typical bronchoscopic findings are bronchial stenosis or obstruction, erythematous mucosa, gelatinous or mucoid secretions, fungating or polypoid masses and mucosal ulceration. The high degree of suspicion is essential as a noninvasive diagnosis with sputum culture is difficult to achieve.

Also to presenting with cough and expectoration, our patient had shortness of breath prior to admission, suggesting that there was airway obstruction that led to dyspnea. When we performed the fiberoptic bronchoscopy, without the pathological evidence, we preliminarily considered the lesion as endotracheal TB or malignancy. A definite diagnosis of pulmonary mucormycosis can only be made with fungal etiology and histopathology results. Sputum culture, used as a simple preliminary diagnostic method, is often negative. The correct diagnosis is usually made by histopathology. Histopathological specimens of lesions can be collected by bronchoscopy biopsy, percutaneous needle biopsy or open lung biopsy; all of which show tissue invasion with characteristic broad, nonseptate hyphae with right-angle branching. As demonstrated by the bronchoscopy biopsy, our patient was diagnosed with confirmed mucormycosis.

Combined surgical and medical treatment of zygomycosis has a reported mortality of about 30%, compared with medical treatment alone which has a mortality of 55%. Furthermore, treatment with liposomal amphotericin B was associated with a 67% survival rate compared with 39% survival with amphotericin B (P = 0.02) among patients with cancer who experienced mucormycosis. Treatment of zygomycosis consists of the prompt administration of amphotericin treatment, reversal of the underlying condition preferentially combined with surgical resection of the necrotic tissue.

Pulmonary mucormycosis is relatively rare disease, with growing number of immunosuppressed patients, it may become more common. Maintaining a high level of suspicion is essential in patients with a pneumonic process, which fails to respond to antibacterial agents, either clinically or radiologically.

 
  References Top

1.
Furbinger P. Observations on lungenmycose beinmeschen. Arch Pathol Anat Physiol Klin Med 1876;66:330-65.  Back to cited text no. 1
    
2.
Garg R, Marak RS, Verma SK, Singh J, Snjay P, Prasad R. Pulmonary mucormycosis mimicking as pulmonary tuberculosis: A case report. Lung India 2008;25:129-31.  Back to cited text no. 2
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3.
Lahiri TK, Agarwal D, Reddy GE, Bajoria A. Pulmonary mucoraceous fungal ball. Indian J Chest Dis Allied Sci 2001;43:107-10.  Back to cited text no. 3
    
4.
Sarkar S, Jash D, Maji A, Maikap MK. Solitary pulmonary nodule: A rare presentation of pulmonary mucormycosis in an immunocompetent adult. Lung India 2014;31:70-2.  Back to cited text no. 4
[PUBMED]  Medknow Journal  
5.
Luo LC, Cheng DY, Zhu H, Shu X, Chen WB. Inflammatory pseudotumoural endotracheal mucormycosis with cartilage damage. Eur Respir Rev 2009;18:186-9.  Back to cited text no. 5
    
6.
Chayakulkeeree M, Ghannoum MA, Perfect JR. Zygomycosis: The re-emerging fungal infection. Eur J Clin Microbiol Infect Dis 2006;25:215-29.  Back to cited text no. 6
    
7.
Bigby TD, Serota ML, Tierney LM Jr, Matthay MA. Clinical spectrum of pulmonary mucormycosis. Chest 1986;89:435-9.  Back to cited text no. 7
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8.
Lee FY, Mossad SB, Adal KA. Pulmonary mucormycosis: The last 30 years. Arch Intern Med 1999;159:1301-9.  Back to cited text no. 8
    
9.
Donohue JF. Endobronchial mucormycosis. Chest 1983;83:585.  Back to cited text no. 9
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10.
Husari AW, Jensen WA, Kirsch CM, Campagna AC, Kagawa FT, Hamed KA, et al. Pulmonary mucormycosis presenting as an endobronchial lesion. Chest 1994;106:1889-91.  Back to cited text no. 10
    


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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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