Mucormycosis in HIV positive diabetic individual amid covidien epoch - A rare case report

Mandavi Waghmare; Anwesha Banerjee; Janice Pinto; Naveen Shetty

doi:10.4103/jiaomr.jiaomr_274_21

CASE REPORT

Year : 2022 | Volume : 34 | Issue : 2 | Page : 237-240

Mucormycosis in HIV positive diabetic individual amid covidien epoch - A rare case report

Mandavi Waghmare, Anwesha Banerjee, Janice Pinto, Naveen Shetty
Department of Oral Medicine and Radiology, D. Y. Patil University-School of Dentistry, Navi Mumbai, Maharashtra, India

Date of Submission	20-Sep-2021
Date of Decision	11-Mar-2022
Date of Acceptance	08-May-2022
Date of Web Publication	22-Jun-2022

Correspondence Address:
Mandavi Waghmare
B7/201, Safal Complex, Plot No 17, Sector 19 A, Nerul, Navi Mumbai, Maharashtra
India

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/jiaomr.jiaomr_274_21

Abstract

Mucormycosis is an opportunistic fulminant fungal infection that can cause significant morbidity and frequent mortality in susceptible patients. Common predisposing factors include diabetes mellitus and immunosuppression. Even though this fungus is ubiquitous, the immune system usually prevents the disease, and it is rare. But in the present pandemic era, Mucormycosis has become a prevalent disease among immunocompromised patients and individuals with systemic pathosis. The infection begins in the nose and paranasal sinuses due to inhalation of fungal spores. The fungus invades the arteries leading to thrombosis that subsequently causes tissue necrosis. The infection can spread to orbital and intracranial structures by direct invasion of blood vessels. Here, we describe an interesting and rare case of sino-nasal mucormycosis in a seropositive, uncontrolled diabetic and suspected Covid positive individual to emphasize early diagnosis and treatment of this fatal fungal infection.

Keywords: AIDS, diabetes mellitus, immunosuppression, mucormycosis, SARS CoV-2 virus infection

How to cite this article:
Waghmare M, Banerjee A, Pinto J, Shetty N. Mucormycosis in HIV positive diabetic individual amid covidien epoch - A rare case report. J Indian Acad Oral Med Radiol 2022;34:237-40

How to cite this URL:
Waghmare M, Banerjee A, Pinto J, Shetty N. Mucormycosis in HIV positive diabetic individual amid covidien epoch - A rare case report. J Indian Acad Oral Med Radiol [serial online] 2022 [cited 2023 Jan 31];34:237-40. Available from: http://www.jiaomr.in/text.asp?2022/34/2/237/347922

Introduction

Mucormycosis, an angioinvasive disease caused by fungi of the order Mucorales, is known to occur in immunocompromised conditions.^[1] The ongoing coronavirus pandemic has seen a sudden surge in this opportunistic infection, thus increasing the rate of morbidity and mortality. In the present crisis, diabetes mellitus (DM) has been the most common risk factor linked with mucormycosis in India, along with careless use of corticosteroids, increased intracellular iron, and transmission via water in an oxygen humidifier.^[2]

HIV-associated mucormycosis is a rarity and is inadequately reported in the literature. Here's reporting a case of mucormycosis in an HIV-positive diabetic individual with SARS CoV-2 Virus infection.

Case Report

Patient information

A 52-year-old male patient, the serviceman by occupation, reported to the department with a chief complaint of mobile teeth in the upper jaw for three days with foul-smelling discharge from the nose and mouth. The patient was a known diabetic for seven years and was on treatment with oral hypoglycaemics.

The history of the present illness revealed that the patient developed fever and cough one month ago. He visited a general physician and was prescribed medications for the same and advised RT- PCR. The patient refrained from doing RT-PCR and started on medication prescribed by the physician. However, he developed an extra-oral swelling and numbness on the right side of the face after five days, for which he approached multiple clinics but did not comply with treatment anywhere. Timeline of case given in [Table 1].

Table 1: Timeline of case

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Clinical findings

On general examination, vitals were normal. On extra-oral examination, facial asymmetry was noted on the right side of the face due to swelling extending from the right lower eyelid to the ala-tragus line with obliteration of the right nasolabial fold. The swelling was soft to firm in consistency on palpation and presented with localized loss of sensation.

Intra-oral examination revealed a localized swelling over the attached gingiva i.r.t 15, 16, 17, and an ulcer palatally i.r.t 16, 17. Inflammation of interdental papilla was seen i.r.t 11, 21. Small localized swelling was observed in 11, 21 [Figure 1]a,[Figure 1]b,[Figure 1]c. The swellings were soft in consistency. Grade I mobility was noted in all maxillary teeth. Based on the clinical examination, a provisional diagnosis of suspected COVID-19 associated mucormycosis of the maxilla was given. With a differential diagnosis of chronic suppurative osteomyelitis (secondary) and generalized periodontitis as a manifestation of systemic disease.

Figure 1: (a) Extra-oral image showing presence of a diffuse swelling involving the right side of the face. (b) Reveals an oval shaped ulcer approximately 0.7mm present palatally i.r.t 16, 17. (c) A well-defined swelling is noted in the buccal vestibule extending from 13 to 17

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Diagnostic assessment

The patient's blood profile on admission is given in [Table 2]. The patient tested positive for HIV on the routine pre-operative investigation. HRCT was done post-hospitalization, which revealed fibrotic changes with adjacent pleural thickening in the anterior segment of the right upper lobe. These findings are consistent with COVID-19 pneumonitis.

Table 2: Investigation chart showing details of blood profile and urine evaluation. (Courtesy: D Y Patil Hospital – Medical Laboratory Report)

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CT and CBVI did a radiographic assessment. CT revealed bilateral opacification of the maxillary sinus, destruction of the right lateral wall of the nasal cavity, and opacification of the right sphenoid sinus and ethmoidal air cells [Figure 2]a and [Figure 2]b.

Figure 2: (a) CT Scan (Coronal section, bone window) shows bilateral opacification of maxillary sinus and destruction of lateral wall of right nasal cavity. (b) CT Scan (Axial section, bone window) shows opacification of right ethmoidal air cells

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CBVI Sagittal section shows irregular areas of bone loss with interspersed areas of bony sclerosis extending from 21 to 17. Loss of buccal and lingual cortical plate noted from 11-17 region. Severe bone loss with 15, 16 causing furcation involvement. Loss of floor of the maxillary sinus and lateral wall and opacification on the right side. The axial section shows discontinuity of the lateral wall of the nasal cavity and posterolateral wall of the right maxillary sinus [Figure 3]a,[Figure 3]b,[Figure 3]c,[Figure 3]d.

Figure 3: (a) CBCT (True-pan Image) shows severe bone loss and furcation involvement i.r.t 15,16,17 with opacification and breach in the lateral wall of right maxillary sinus. (b) CBCT (Coronal section) showing bony erosion i.r.t 15 and loss of buccal and palatal cortical plate and discontinuity of floor and facial wall of maxillary sinus. (c) CBCT (Axial section) showing irregular bone loss extending from 21 to 27 and loss of buccal and palatal cortical plate noted. (Moth eaten appearance) (d) CBCT (Axial section) showing opacification of sinus and right nasal cavity with loss of lateral wall of maxillary sinus

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On PAS stain examination, an aspirate from the right maxillary bone showed broad aseptate fungal hyphae, suggestive of mucormycosis [Figure 4]. Based on all the findings, the final diagnosis was mucormycosis associated with osteomyelitis of the maxilla in a diabetic HIV-positive patient.

Figure 4: (100x magnification on Electron Microscope)- Histopathological evaluation shows fungal hyphae on PAS staining

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Therapeutic intervention

Surgical debridement of the sino-nasal complex and maxillectomy was performed under general anesthesia. The patient was administered liposomal Amphotericin-B 295 mg in 200 ml 5% dextrose intravenously once daily for five days.

Outcome

On the fifth day post-surgery, the patient passed away due to septic shock with multi-organ dysfunction syndrome and acute renal failure.

Discussion

India has the highest burden of mucormycosis worldwide, with an estimated prevalence of 140 cases per million population.^[3] DM is the single common risk factor for mucormycosis in India, reported in 54-76% of cases, and HIV is reported in 1.5% of cases.^[1] In the present pandemic, mucormycosis emerged as an imminent threat, with 14872 cases reported from the beginning till May 19, 2021.^[4]

Hyperglycemia decreases the phagocytic ability of leukocytes and helps the fungal spore to thrive and increase. DK also temporarily disrupts the ability of transferrin to bind iron, thus eliminating the host defense mechanism.^[5],[6]

COVID-19 infection induces hyperglycemia by decreasing insulin secretion and developing insulin resistance, leading to the growth of invasive mucormycosis.^[7] It also causes immune dysregulation with fewer T lymphocytes and increases interleukin and tumor necrosis factors.^[8]

HIV though not adequately reported, is also a risk factor in mucormycosis. Low CD4 counts (cells <50/mm³), neutropenia, and intravenous drug users are predisposing factors for mucormycosis. In a systematic review of 929 cases of mucormycosis, co-infection of HIV is reported in just 2% of cases.^[9]

Imaging helps assess the extent of the disease and is indispensable for surgical planning. This is the first case presenting CBCT images in mucormycosis showing rarefaction, erosion, and permeative destruction of the maxillary bone, suggestive of mucormycosis-induced osteomyelitis.

Though CT and MRI are considered the gold standard in diagnosing mucormycosis, CBCT provides a low dose, low-cost alternative to conventional CT and plays a pivotal role in detecting the early involvement of paranasal sinuses and the surrounding structures.

This is a unique case of mucormycosis in the Covidien era with a rare trilogy of Diabetes, HIV, and SARS-CoV-2 infection in a case of Rhino-Cerebral Mucormycosis. However, the patient succumbed to the disseminated infection, which could have been prevented with timely intervention.

Note

This is the first case presenting with a trilogy of Diabetes, AIDS, and SARS CoV-2 infection in a patient with mucormycosis.

Though medical literature suggests CT and MRI as the gold standard in diagnosing mucormycosis, this case also highlights the importance of CBCT as an important diagnostic aid for an oral physician in diagnosing mucormycosis.

Patient perspective

Complications during the treatment phase led to the demise of the patient.

Conclusion

Early diagnosis and treatment of mucormycosis are extremely important. Regular health check-ups among the Indian population should be readily emphasized to diagnose and treat underlying diseases and control morbidity and mortality.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. The patient(s) has/have given his/her/their images and other clinical information to be reported to the journal. The patient understands that his name and initials will not be published, and due efforts will be made to conceal his identity.

Acknowledgments

We express our heartfelt gratitude to Dr. Swati Gotmare, Professor, Department of Oral and Maxillofacial Pathology for her contribution towards histopathological contribution.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Nallapu V, Vuppalapati HB, Sambhana S, Balasankulu B. Rhinocerebral mucormycosis: A report of two cases. J Indian Acad Oral Med Radiol 2015;27:147-51. [Full text]
2.	Gupta A, Sharma A, Chakrabarti A. The emergence of post covid -19 mucormycosis in India. Can we prevent it? Indian J Ophthalmol 2021;69:1645-47. [PUBMED] [Full text]
3.	Mohanty N, Misra SR, Sahu SR, Mishra S, Vasudevan V, Kailasam S. Rhinomaxillary mucormycosis masquerading as chronic osteomyelitis- A series of four rare cases with review of the literature. J Indian Acad Oral Med Radiol 2012;24:315-23. [Full text]
4.	Raut A, Huy NT. Rising incidence of mucormycosis in patients with COVID-19: Another challenge for India amidst the second wave? Lancet 2021;9:e77.
5.	Artis WM, Fountain JA, Delcher HK, Jones HE. A mechanism of susceptibility to mucormycosis in diabetic ketoacidosis. Transferrin and iron availability. Diabetes 1982;31:1109-14.
6.	Marx RF, Stern D. Inflammatory, Reactive and Infectious Disease in Oral and Maxillofacial Pathology. In: Carol Stream III editor. Textbook of Oral and Maxillofacial Pathology: a rationale for diagnosis and treatment. 2^nd ed. USA: Quintessence Publishing; 2003. p.104-6.
7.	Hussain A, Bhowmik B, do Vale Moreira NC. COVID-19 and diabetes: Knowledge in progress. Diabetes Res Clin Pract 2020;162:108142.
8.	Gangneux JP, Bougnoux ME, Dannaoui E, Cornet M, Zahar JR. Invasive fungal disease during covid 19, we should be prepared. J Mycol Med 2020;30:100971. doi: 10.1016/j.mycmed. 2020.100971.
9.	Roden MM, Zoutis TE, Buchanan WL, Knudsen TA, Sarkisova TA, Schaufele RL. Epidemiology and outcome of zygomycosis: A review of 929 reported cases. Clin Infect Dis 2005;41:634-53.