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| CASE REPORT |
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| Year : 2023 | Volume
: 6
| Issue : 2 | Page : 76-80 |
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Isolated pulmonary mucormycosis in diabetic patients
Vyshnavi Rajeev, Ajay Narasimhan, Jebin Roger, R Narasimhan
Department of Respiratory Medicine, Apollo Main Hospital, Chennai, Tamil Nadu, India
| Date of Submission | 31-Mar-2023 |
| Date of Decision | 26-Apr-2023 |
| Date of Acceptance | 27-Apr-2023 |
| Date of Web Publication | 13-Jul-2023 |
Correspondence Address:
Dr. Vyshnavi Rajeev
Apollo Main Hospital, Greams Road, Chennai - 600 006, Tamil Nadu
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/japt.japt_14_23
Mucormycosis refers to the infection caused by a growing number of members of the Mucorales. Mucormycosis has emerged as an important opportunistic infection in severely immunocompromised patients with hematological malignancies and recipients of stem cell or organ transplantation and in poorly controlled diabetics. The diagnosis of mucormycosis relies upon histopathology and culture. Blood tests are of limited diagnostic value. Here, we present two cases of mucormycosis in diabetic patients diagnosed with tissue biopsy.
Keywords: GeneXpert MTB, mucormycosis, Video-assisted thoracic surgery left upper lobectomy
How to cite this article:
Rajeev V, Narasimhan A, Roger J, Narasimhan R. Isolated pulmonary mucormycosis in diabetic patients. J Assoc Pulmonologist Tamilnadu 2023;6:76-80 |
How to cite this URL:
Rajeev V, Narasimhan A, Roger J, Narasimhan R. Isolated pulmonary mucormycosis in diabetic patients. J Assoc Pulmonologist Tamilnadu [serial online] 2023 [cited 2023 Sep 30];6:76-80. Available from: https://www.japt.in//text.asp?2023/6/2/76/381413 |
| Introduction | |  |
Mucormycosis is an invasive fungal disease that is relatively rare but often fatal and rapidly progressive. Maintaining a high level of suspicion is important in any patient in the right clinical setting with a pneumonic process that fails to respond to antibacterial agents, either clinically or radiologically. The diagnosis is rarely obtained by cultures because of processing in microbiology laboratories, and more aggressive bronchoscopic or surgical approaches should be pursued to obtain histopathologic specimens.
| Case Series | | |
Case 1
A 65-year-old woman presented to the outpatient department with history of intermittent low-grade fever and dry cough for 1 month. She was a known case of diabetes and hypertension on regular medications.
On general examination, she was moderately built, afebrile, and maintained optimal saturation with room air. On systemic examination, she had decreased breath sounds over the left upper hemithorax with normal vesicular breath sounds over other areas.
Laboratory tests showed total counts of 13,130 with neutrophils at 70% and lymphocytes at 26%, hemoglobin at 10.3 g%, and HbA1c of 9.1%.
Computed tomography (CT) chest axial view [Figure 1] and coronal view [Figure 2] showed abrupt cutoff of left upper lobe bronchus with collapse consolidation of left upper lobe, mucoid impaction of left upper lobe bronchus, suggestive of the obstructive endobronchial lesion and mediastinal lymphadenopathy. | Figure 1: CT chest axial view showing collapse consolidation of left upper lobe. CT: Computed tomography
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 | Figure 2: CT chest coronal view showing abrupt cut off of left upper lobe bronchus with collapse consolidation of left upper lobe. CT: Computed tomography
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She underwent a bronchoscopy [Figure 3] and [Figure 4] which showed mass encasing the left main bronchus and left upper lobe bronchi which was highly vascular. Bronchial wash and bronchial brush cytology were negative for malignancy. Stains, cultures, and GeneXpert MTB were also negative.
CT-guided biopsy was done which showed suppurative inflammation with clusters of foamy histiocytes and a vague epitheliod granuloma with a possibility of tuberculosis.
Positron emission tomography-CT whole body [Figure 5] showed mildly fluorodeoxyglucose (FDG) avid heterogeneously enhancing ill-defined soft-tissue density lesion within the left upper lobe and hypermetabolic subcarinal and left paratracheal nodes. | Figure 5: PET-CT showing mildly FDG avid heterogenously enhancing ill-defined soft-tissue density lesion within the left upper lobe. Arrow shows enlarged left para-aortic lymph node. PET-CT: Positron emission tomography-computed tomography, FDG: Fluorodeoxyglucose
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She underwent left upper lobectomy sleeve resection and lung tissue fungal staining showed many aseptate ribbon-like hyphae, suggestive of mucormycosis. Tissue biopsy also confirmed the diagnosis of mucormycosis. Treatment was started initially with amphotericin B injection for which she developed adverse reactions and was changed to oral posaconazole and she was discharged after 1 week of surgery. She improved clinically when she presented to the outpatient department after 1 month for follow-up.
Case 2
A 52-year-old man presented to the emergency department with history of low-grade intermittent fever, cough with mucopurulent sputum, and weight loss for 1 month. History of one episode of blood-tinged sputum was present 2 weeks back from the time of presentation. History of chest pain while coughing and breathlessness was also present for 2 weeks.
He was diagnosed with uncontrolled diabetes 1 month ago and was on treatment with insulin injection at the time of presentation. He had uncontrolled asthma and was not a smoker.
On general examination, he was moderately built, febrile, had tachycardia, and maintained optimal saturation in room air.
On systemic examination, there was bronchial breath sounds over the left upper hemithorax with normal vesicular breath sounds and bilateral wheeze over all other areas.
Laboratory investigations showed elevated urea, creatinine, and INR values.
Chest X-ray [Figure 6] showed bilateral upper and middle zone cystic shadows. CT chest axial [Figure 7] and [Figure 8] and coronal views [Figure 9] and [Figure 10] showed bilateral upper lobe thick-walled cavities. | Figure 6: Chest X-ray showing bilateral upper and middle zone cystic shadows
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 | Figure 7: CT chest axial view showing right upper lobe cavity. CT: Computed tomography
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 | Figure 8: CT chest axial view showing left upper lobe cavity. CT: Computed tomography
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 | Figure 9: CT chest coronal view showing right upper lobe cavity. CT: Computed tomography
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 | Figure 10: CT chest coronal view showing left upper lobe cavity. CT: Computed tomography
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Bronchoscopy was done after correcting INR and it showed mucosal edema in the left upper lobe and lingular bronchi [Figure 11] and purulent secretions in the right upper lobe apical segmental bronchus [Figure 12]. | Figure 11: Bronchoscopy showing mucosal edema in left upper lobe and lingular bronchi
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 | Figure 12: Bronchoscopy showing purulent secretions in right upper lobe apical segmental bronchus
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Bronchial wash and tissue sample fungal stain showed aseptate and septate hyphae, suggestive of mucormycosis. Transbronchial lung biopsy histopathological examination showed bronchiolitis obliterans organizing pneumonia-like changes with fungal ball mixed Aspergillus and Mucor. Gram stain, AFB stain, GeneXpert MTB, and cytology were negative.
Treatment was treated initially with amphotericin B injection. Cardiovascular and thoracic surgery opinion was obtained and it was decided to perform video-assisted thoracic surgery left upper lobectomy. During surgery, it was found that the disease segment crossed the fissural line, so the decision was made to proceed with an open left pneumonectomy.
He developed refractory septic shock on 5th postoperative day in association with multidrug-resistant tuberculosis, Klebsiella pneumonia, and surgical site infection, followed by left stump leak. However, despite escalating treatment, he died on the 12th postoperative day.
| Discussion | | |
Mucormycosis is an invasive fungal disease that is relatively rare but often fatal and rapidly progressive. The incidence of mucormycosis has recently been increasing. It is the second most common invasive fungal disease, ranking second only to aspergillosis. The genera that cause most cases of mucormycosis are Rhizopus, Mucor, and Lichtheimia (previously Absidia). Common predisposing factors include hematological malignancy, diabetes, transplantation, use of corticosteroids or immunosuppressants, and trauma. Common clinical types of mucormycosis include rhino cerebral (27%–39%), pulmonary (20%–30%), dermal (19%–26%), disseminated (3%–15%), and gastrointestinal types (7%–8.5%). Pulmonary mucormycosis (PM) is the second or third most common clinical subtype of mucormycosis.[1]
Most human infections result from inhalation of fungal sporangiospores that have been released in the air or direct inoculation of organisms into disrupted skin or mucosa. The Mucorales are ubiquitous in nature, but their precise ecology remains to be determined; they are thermotolerant and are usually found in decaying organic matter.[2]
Symptoms of PM are nonspecific and include fever, cough, dyspnea, and chest pain. Lesions typically involve the parenchyma but may extend into the chest wall, pulmonary artery, aorta, mediastinum, or pericardium. Hemoptysis can be caused by the invasion of pulmonary arteries.[3]
Maintaining a high level of suspicion is important in any patient in the right clinical setting with a pneumonic process that fails to respond to antibacterial agents, either clinically or radiologically. In some patients, an apparent initial improvement is followed by recrudescence of symptoms shortly after, maybe due to a transient response of a postobstructive bacterial pneumonitis secondary to endobronchial disease. These scenarios should heighten concerns about the possibility of fungal infection. The diagnosis is rarely obtained by cultures because of processing in microbiology laboratories, and more aggressive bronchoscopic or surgical approaches should be pursued to obtain histopathologic specimens.[4]
Successful management of mucormycosis is based on a multimodal approach, including reversal or discontinuation of underlying predisposing factors (if possible), early administration of active antifungal agents at the optimal dose, complete removal of all infected tissues, and the use of various adjunctive therapies. Rapid correction of metabolic abnormalities is mandatory in patients with uncontrolled diabetes and suspected of mucormycosis.[5]
The recommended antifungal agent is liposomal amphotericin B, but there are concerns about the limited penetration of antifungals to the affected tissues, due to the substantial necrosis that accompanies this infection. For this reason, surgical debridement has been advocated. Several studies have shown that the combination of early surgical resection and antifungal therapy has a significant improvement in survival when compared to antifungal therapy alone. This early aggressive approach is critical, since the delay of effective systemic antifungal treatment after the diagnosis of mucormycosis may increase mortality. Therefore, unless surgery is contraindicated, the recommended therapy for PM is early surgical debridement in conjunction with early amphotericin B therapy (6 d after diagnosis). The duration of therapy is individualized to the patient, but the near normalization of radiographic abnormalities, the negativity of cultures, or the resolution of the immunosuppressed state can be used as surrogates to stop therapy.[6]
Both of our patients presented with nonresolving pneumonia, in which mucormycosis was diagnosed with tissue biopsy and was treated with surgical resection and antifungals. These cases are being reported because of the need to suspect mucormycosis in nonresolving pneumonia, especially in diabetic patients.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the patients have given their consent for their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity and preserve anonymity.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Peng M, Meng H, Sun Y, Xiao Y, Zhang H, Lv K, et al. Clinical features of pulmonary mucormycosis in patients with different immune status. J Thorac Dis 2019;11:5042-52.  |
| 2. | Petrikkos G, Skiada A, Lortholary O, Roilides E, Walsh TJ, Kontoyiannis DP. Epidemiology and clinical manifestations of mucormycosis. Clin Infect Dis 2012;54 Suppl 1:S23-34. |
| 3. | Serris A, Danion F, Lanternier F. Disease entities in mucormycosis. J Fungi (Basel) 2019;5:23. |
| 4. | Lee FY, Mossad SB, Adal KA. Pulmonary mucormycosis: The last 30 years. Arch Intern Med 1999;159:1301-9. |
| 5. | Skiada A, Lass-Floerl C, Klimko N, Ibrahim A, Roilides E, Petrikkos G. Challenges in the diagnosis and treatment of mucormycosis. Med Mycol 2018;56:93-101. |
| 6. | Fernandez JF, Maselli DJ, Simpson T, Restrepo MI. Pulmonary mucormycosis: What is the best strategy for therapy? Respir Care 2013;58:e60-3. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12]
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