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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 10  |  Issue : 1  |  Page : 43-45

Pulmonary mucormycosis: a rare case report


Himalayan Institute of Medical Sciences, Swami Rama Himalayan University, Jolly Grant, Dehradun, Uttarakhand, India

Date of Submission21-Feb-2021
Date of Acceptance20-Nov-2021
Date of Web Publication19-Apr-2022

Correspondence Address:
Dr. Varuna Jethani
Himalayan Institute of Medical Science, Jolly Grant, Dehradun 248140, Uttarakhand
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jacp.jacp_7_21

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  Abstract 


Mucormycosis is a rare fungal infection caused by Mucorales order fungi. It is observed in patients with hematologic malignancies, diabetes mellitus, and immunocompromised states. Clinical diagnosis is difficult in pulmonary mucormycosis, and early diagnosis is needed for this life-threatening infection. We report a case of pulmonary mucormycosis in a 52-year-old male with type 2 diabetes diagnosed on bronchoscopic-guided biopsy. We aim to illustrate the need for a high clinical suspicion for the diagnosis of mucormycosis and to report the importance of early and aggressive initiation of antifungal therapy.

Keywords: Amphotericin, cavity, fatal, mucormycosis


How to cite this article:
Sharma D, Jethani V, Pant S, Joshi S. Pulmonary mucormycosis: a rare case report. J Assoc Chest Physicians 2022;10:43-5

How to cite this URL:
Sharma D, Jethani V, Pant S, Joshi S. Pulmonary mucormycosis: a rare case report. J Assoc Chest Physicians [serial online] 2022 [cited 2022 Sep 25];10:43-5. Available from: https://www.jacpjournal.org/text.asp?2022/10/1/43/339695




  Introduction Top


Pulmonary mucormycosis is rare but fatal infection caused by the genera Mucorales which occurs after inhalation of spores into the bronchioles and alveoli.[1] There are six forms of mucormycosis which have been commonly reported which include rhinocerebral, pulmonary, cutaneous, gastrointestinal, disseminated, and uncommon presentations.[2],[3] Incidence of pulmonary mucormycosis is 25% as reported in literature in comparison with other forms.[4] In immunocompetent host, they do not cause disease but as soon as the immunity decreases due to any cause, disease manifestation occurs and causes invasive disease. There are no reliable serologic, polymerase chain reaction (PCR)-based, or skin tests for mucormycosis and diagnosis relies upon the histopathology with identification of broad, nonseptate hyphae which branch at 90° in tissue with culture confirmation.[5]

In this study, we present a rare case of pulmonary mucormycosis in patient of type 2 diabetes mellitus.


  Case summary Top


A 52-year-old male known diabetic on irregular oral hypoglycemic presented in emergency with complaints of right-sided chest pain diffuse dull aching radiating to back for 3 months associated with fever and hemoptysis daily three to four teaspoons for 10 days. Fever was documented on and off around 37.2°C to 37.7°C not associated with chills and rigor. On admission, the patient was stable. General physical examination was unremarkable. Local examination revealed crepts on the right side of chest. Laboratory parameters were suggestive of mild anemia (10 g/dL), raised blood sugar level (455 mg/dL), and high HbA1c (10%) which was managed with insulin. Nasopharyngeal swab for reverse transcription-PCR of severe acute respiratory syndrome coronavirus 2 was negative. Chest X-ray revealed thick-walled cavitatory lesion in mid and lower zones on the right side [Figure 1] raising suspicion of tuberculosis as being common in India. Initial sputum gram stain, acid fast bacilli (AFB), and potassium hydroxide (KOH) mount were negative. Contrast-enhanced computed tomography (CECT) thorax was performed which showed multiple thick-walled cavitatory lesions on right side with normal great vessel [Figure 2]. Patient was then taken up for bronchoscopy which showed mucopurulent secretions coming out from right upper lobe bronchus with mucosal swelling. Bronchoalveolar lavage (BAL) was taken from right upper lobe which grew Burkholderia in culture, sensitive to cotrimoxazole and clindamycin and rest BAL sample came out to be negative for AFB, KOH mount, nocardia, pneumocystis, gram stain, and cytology.
Figure 1 Chest X-ray: thick-walled cavitatory lesion in mid and lower zones on the right side.

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Figure 2 Contrast-enhanced computed tomography thorax: multiple thick wall cavitatory lesions on right side with normal great vessel.

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Biopsy from mucosal swelling was suggestive of acute necrotizing inflammation with fungal hyphae of mucormycosis positive for Gomori methenamine silver stain [Figure 3].
Figure 3 Histopathology slides: acute necrotizing inflammation with fungal hyphae of mucormycosis.

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Patient was started on liposomal amphotericin B at the dose of 5 mg/kg which was increased to 10 mg/kg along with intravenous antibiotics − clindamycin (600 mg thrice daily intravenous) and cotrimoxazole as per culture report and other symptomatic management. During hospital stay, patient had episode of massive hemoptysis for which he was shifted to critical care unit, managed conservatively, and transfused blood later shifted back to general ward as hemoptysis settled.

Patient gradually improved symptomatically as fever and hemoptysis settled. Repeat chest X-ray and CT thorax was carried out which showed marked reduction in size of cavitatory lesions. Repeat bronchoscopy was performed which showed only erythematic mucosa without any secretions and mucosal swelling resolved. Repeat mucosal biopsy was taken which was suggestive of chronic inflammation and did not show fungal hyphae. Liposomal amphotericin B treatment was given for 46 days with monitoring of kidney function test.


  Discussion Top


Pulmonary mucormycosis is an uncommon fungal infection that has very high morbidity and mortality. The high mortality can be attributable to the delay it carries in making the diagnosis.[6]

The hyphae of these Mucorales are different as they are broad, wide, thick walled being 5 to 15 μ in diameter, ribbon-like, aseptate hyphae that branch at right angles. The absence of regular septations might be the cause of fragile nature of the hyphae and the difficulty of growing the agents of mucormycosis from clinical specimens. Ketone reductase is an enzyme which allows them to live in high glucose and acidic conditions; hence, patient with diabetic ketoacidosis in which there is macrophage dysfunction their serum promotes the growth of Rhizopus.[7]

Rhino-orbital-cerebral and pulmonary mucormycosis are acquired by the inhalation of these spores. In healthy individuals, cilia push back these spores to the pharynx and they get cleared. In susceptible individuals, infection usually begins in the nasal turbinates or the alveoli. Mucormycosis being angio-invasive in nature; thus, infarction of infected tissues is pathologic of invasive disease.[8]

The risk factors of mucormycosis are immunocompromised states such as diabetes mellitus, on chronic steroid therapy, hematologic malignancies, solid organ transplant patients, patients on deferoxamine therapy, intravenous drug abusers, burns, trauma, and malnutrition.[5] In pulmonary mucormycosis, the most common underlying immunocompromised states that have been reported in literature are hematologic malignancies followed by patients on chronic glucocorticoid therapy and deferoxamine followed by solid organ transplantation. In diabetics, rhino-cerebral mucormycoses have been reported to be more common.[9] In our case, patient was diabetic with poor control presented with fever and hemoptysis but without ketoacidosis as reported in literature as an add-on risk factor in diabetes mellitus. It is very important to differentiate between pulmonary mucormycosis and invasive pulmonary aspergillosis that is caused by another fungal entity as both have almost similar presentation with history of being in immunocompromised host and early radiologic and histopathologic samples help to differentiate between two and further early initiation of treatment as these conditions have high mortality rate. Radiologic findings that have been commonly reported in literature for pulmonary mucormycosis are nodules, focal consolidation, mass, pleural effusion, bird nest sign, and reverse halo sign, and comparing it with invasive pulmonary aspergillosis, the important radiologic features are peribronchial consolidation, bronchial wall thickening, air crescent sign, and halo sign.[10],[11] Infarction with cavity lesion and air crescent sign are uncommon and less reported in literature but in our case, the main radiologic finding was multiple cavitatory lesions. These cavitatory lesions might mimic tuberculosis being common in India but a keen observation of endobronchial findings can prompt for pulmonary mucormycosis as we did in our case.

As per revised criteria of the European Organization for Research and Treatment of Cancer/Mycosis Study Group, pulmonary mucormycosis was defined by histologic evidence of tissue invasion consisting of nonseptated, right-angle branching filamentous fungi plus recovery of Mucorale species by culture of specimens from pulmonary tissue or positivity for immunohistochemical staining with anti-Rhizopus arrhizus monoclonal antibody (LSBio, Seattle, WA, USA). Probable pulmonary mucormycosis was defined as the presence of host factors together with one or more clinical indications, such as dense, well-circumscribed lesions with or without a halo sign, and an air-crescent sign or cavity on CT, and mycologic evidence of Zygomycetes in sputum or BAL fluid culture.[11]

The duration of therapy of liposomal amphotericin B has to be individualized depending upon clinicoradiologic improvement. In our case, it was 46 days and was stopped as patient was clinically and radiologically better and bronchoscopic findings had resolved. Patient is doing well and is on regular follow-up. No new cavity or lesion or worsening was observed so far.


  Conclusion Top


Pulmonary mucormycosis should be considered in mind in cases of cavitary lung diseases in immunocompromised patients to prevent delay in treatment and thereby decreasing morbidity and mortality.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Spellberg B, Maertens J. Mucormycosis. In: Safdar A, ed. Principles and Practice of Transplant Infectious Diseases. New York, NY: Springer 2019. pp. 1-4.  Back to cited text no. 1
    
2.
Pennell E, Pecson I, Nakamura C, Galvis AE. Pulmonary mucormycosis in an adolescent female with type 1 diabetes mellitus. IDCases 2018;14:00474.  Back to cited text no. 2
    
3.
Serris A, Danion F, Lanternier F. Disease entities in mucormycosis. J Fungi 2019;5:23.  Back to cited text no. 3
    
4.
Luo Z, Zhang L. Diagnosis and treatment of pulmonary mucormycosis: a case report. Exp Ther Med 2017;14:3788-91.  Back to cited text no. 4
    
5.
Walsh TJ, Gamaletsou MN, McGinnis MR, Hayden RT, Kontoyiannis DP. Early clinical and laboratory diagnosis of invasive pulmonary, extrapulmonary, and disseminated mucormycosis (Zygomycosis). Clin Infect Dis 2012;54(Suppl 1):S55-60.  Back to cited text no. 5
    
6.
Iqbal N, Irfan M, Jabeen K, Kazmi MM, Tariq MU. Chronic pulmonary mucormycosis: an emerging fungal infection in diabetes mellitus. J Thorac Dis 2017;9:E121-5.  Back to cited text no. 6
    
7.
Guarner J, Brandt E. Histopathologic diagnosis of fungal infections in the 21st century. Clin Microbiol Rev 2011;24:247-80.  Back to cited text no. 7
    
8.
Green JP, Karras DJ. Commentary. Ann Emerg Med 2012;59:54-5.  Back to cited text no. 8
    
9.
Skiada A, Lass-Floerl C, Klimko N, Ibrahim A, Roilides E, Petrikkos G. Challenges in the diagnosis and treatment of mucormycosis. Med Mycol 2018; 56(Suppl 1): S93-S101.  Back to cited text no. 9
    
10.
McAdams HP, Rosado de Christenson M, Strollo DC, Patz Jr EF. Pulmonary mucormycosis: radiologic findings in 32 cases. Am J Roentgenol 1997;168:1541-8.  Back to cited text no. 10
    
11.
Jung J, Kim MY, Lee HJ et al. Comparison of computed tomographic findings in pulmonary mucormycosis and invasive pulmonary aspergillosis. Clin Microbiol Infect 2015;21:684.e11-8.  Back to cited text no. 11
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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