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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 8  |  Issue : 2  |  Page : 141-145

Mucormycosis: A case report of black fungus as COVID-19 complication


1 Department of Oral Medicine and Radiology, Babu Banarsi Das University of Dental Sciences, Lucknow, Uttar Pradesh, India
2 Department of Internal Medicine, Singhania University, Jhunjhunu, Rajasthan, India

Date of Submission17-Jan-2022
Date of Decision11-Feb-2022
Date of Acceptance04-Mar-2022
Date of Web Publication2-Sep-2022

Correspondence Address:
Prarthana Govil
Department of Oral Medicine and Radiology, Babu Banarsi Das College of Dental Sciences, Lucknow Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jigims.jigims_4_22

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  Abstract 


We report a case of post-COVID-19 mucormycosis infection caused by Rhizopus oryzae. The patient was diagnosed with COVID-19 and treated according to the existing protocols. Following recovery, the patient developed swelling and pain in the right upper region of the face. A diagnosis of mucormycosis was established after cone-beam computed tomography findings. The patient was referred to a government hospital where she was treated according to the COVID protocol. Generally, mucormycosis develops in patients who are immunocompromised or suffering from debilitating disease which was COVID infection and uncontrolled diabetes in this case. The mold usually gains entry through the respiratory tract involving the nose and sinuses, which can further progress to intracranial structures. Early diagnosis and intervention are the keys to good prognosis and decreasing morbidity. Judicious use of drugs is required in COVID-19 patients, especially in immunocompromised patients as research is still in progress. Opportunistic fungal coinfections are one of the complications which are needed to be taken care of. Prophylactic treatment protocols need to be established, along with rational use of corticosteroids.

Keywords: Complication, COVID-19, mucormycosis


How to cite this article:
Govil P, Singh P, Rai P, Tayal A. Mucormycosis: A case report of black fungus as COVID-19 complication. J Indira Gandhi Inst Med Sci 2022;8:141-5

How to cite this URL:
Govil P, Singh P, Rai P, Tayal A. Mucormycosis: A case report of black fungus as COVID-19 complication. J Indira Gandhi Inst Med Sci [serial online] 2022 [cited 2022 Nov 28];8:141-5. Available from: http://www.jigims.co.in/text.asp?2022/8/2/141/355324




  Introduction Top


Mucormycosis, also known as phycomycosis or zygomycosis, is an acute opportunistic infection caused by saprophytic fungus found in soil, bread molds, and decaying fruits and vegetables. The organism can also be cultured from the oral cavity, nasal pharynges, throat, and stool of healthy persons.[1] The disease manifests as rhinocerebral, cutaneous, gastrointestinal, and pulmonary or a disseminated form. Occasional cases arise in the central nervous system or cardiovascular system or follow surgical procedures or severe burns.[2],[3]

Innumerable predisposing risk factors are associated with mucormycosis, and most cases have history of poorly controlled ketoacidosis diabetes, long-term use of antibiotics or steroids or in patients with hematologic malignant conditions.[4],[5] Rarely, a healthy individual with well-controlled diabetics or patients with no underlying immunodeficiency disorder may develop fulminant fungal infection.

The current wave of COVID-19 has resulted in immunocompromised conditions in number of patients, especially one with comorbidities. The use of cocktail of drug therapy had led to the increase in the number of COVID-associated mucormycosis cases all over India. Involvement of the oral cavity usually appears as ulceration of the palate, which results due to invasion of palatal vessel or necrosis. Patients often exhibit facial cellulitis, paresthesia, and anesthesia. Patient also complains of nasal discharge, necrotic turbinates, fever, headache, and lethargy. Without appropriate treatment, the disease spreads into the orbit and brain and results in death.


  Case Report Top


A 40-year-old female patient reported in the Department of Oral Medicine and Maxillofacial Radiology complained of pain and swelling in the right upper region of the face for 1 month.

The patient was apparently asymptomatic 1 month ago. Following that the patient started noticing swelling and feeling pain of gradual onset, continuous in nature, and mild in intensity. The patient took medication for the first 3 days but was not relieved and pain progressed to severe intensity accompanied by fever, so the patient was admitted to the government hospital for 5 days. The patient was still not relieved so took discharge. The patient consulted many doctors and took medication but was not relieved, so after 15 days, the patient got the maxillary second premolar and first molar extracted from a quack. Apparently, following the extraction, the socket never healed completely. The patient was still not relieved. Swelling increased gradually for3 days after initial pain and progressed to attain the present state. The patient was on medication after extraction. The patient was not relieved so was referred to our dental hospital for further evaluation. The patient gave a medical history of diabetes and also gave history of COVID positive 1.5 month ago. The patient was admitted for 14 days and was on oxygen support for 5 days and had taken steroids for 10 days during the tenure of being COVID positive. Extraorally, a diffuse swelling was present in the mid-facial region of the face extending mesio-laterally from the right ala of the nose to 1 cm away from tragus of the ear and supero-inferiorly from periorbital area to 1 cm below ala tragus line measuring approximately 4 cm × 5 cm. The overlying skin was shining and stretched with obliteration of nasolabial fold. All inspector findings were confirmed on palpation. The swelling was tender on palpations. The margins were ill defined with a slight rise in temperature [Figure 1], [Figure 2], [Figure 3].
Figure 1: Extraoral photograph showing asymmetrical face

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Figure 2: Extraoral photograph showing diffuse swelling on the right middle third of the face

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Figure 3: Extraoral photograph showing no deformity on the left side of the face

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Intraorally, a diffuse swelling could be well appreciated in the right maxillary region extending mesio-laterally from the region of 13–18 and supero-inferiorly from 1 cm away from mid-palatal raphe to buccal cortical plate. Unhealed socket could also be inspected from 13 to 17 and was erythematous. The patient while trying to assess intraorally was found to have restricted mouth opening of 13 mm. On palpation, the lesion was tender and firm. The maxillary bone section on the right side was mobile. The patient was advised cone-beam computed tomography which was displayed [Figure 4].
Figure 4: Intraoral photograph showing unhealed lesion on the right maxillary area

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Axial view

In the middle one-third region, a mixed radiolucent and radiopaque lesion could be appreciated which extended from 14 to maxillary tuberosity on the right side, buccolingually from lingual cortex to buccal cortex. The outer and inner cortex were discontinuous and interrupted which extended to zygomatic arch [Figure 5], [Figure 6].
Figure 5: CBCT image showing diffuse radiolucency in the right maxillary area

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Figure 6: CBCT image showing nasal floor discontinuity

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Coronal view

Bilaterally mixed radiolucent radiopaque lesion could be appreciated in the maxillary sinus area extending from apex of maxillary roots to the floor of maxillary sinus and mesio-laterally from medial wall to lateral wall of maxillary sinus [Figure 7].
Figure 7: CBCT image showing radiolucent radiopaque lesion

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Sagittal view

Mixed radiolucent radiopaque lesion could be appreciated in the maxillary sinus area extending supero-inferiorly from apex of tooth 16, 17, and 18 to the floor of maxillary sinus area. The lateral and medial walls of maxillary sinus are discontinuous [Figure 8].
Figure 8: CBCT image showing mixed lesion in maxillary sinus

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The patient was advised incisional biopsy and microscopically, the section from soft tissue revealed tissue bits lined by hyperplastic stratified squamous epithelium with subepithelial tissue showing granulation tissue and dense mixed inflammation. The inflammatory cells comprised lymphocytes, plasma cells, polymorphonuclear cells, few eosinophils, and histiocytes. Sections from bony tissue piece showed bony trabeculae with dense plasma cell-rich mixed inflammatory infiltrate in inter trabecular area. Few hematoxylin-stained broad nonseptate fungal hyphae were noted and areas of hemorrhage were seen. There was however no evidence of malignancy. Correlating the clinical, radiological, and histopathological findings, the diagnosis of fungal infection with granulation tissue was made.

The patient was then referred to a government hospital where she underwent the necessary treatment.


  Discussion Top


The estimated prevalence of mucormycosis is around 70 times higher in India than that in global data. The computational model-based method estimated a prevalence of 14 cases/100,000 individuals in India.[6] Mucormycosis is an invasive fungal infection first described by Paulltauf in 1885.[3] The causative agents are the filamentous fungi of the Mucoraceae family.[2],[3] The most frequently isolated species is Rhizopus oryzae followed by Rhizopus microsporus, and Absidia corymbifera.[2] Rhinocerebral mucormycosis is also referred to as rhino-orbito-cerebral mucormycosis in the respective literature to denote the involvement of the orbital structures.[4] Mucormycosis is one of the most rapidly progressing and fulminant forms of fungal infection which usually begins in the nose and paranasal sinuses following the inhalation of fungal spores. Our patient's infection was apparently induced by COVID-19 complication and tooth extraction. There have been several other reports of rhinocerebral mucormycosis occurring after dental extractions.[3] Once inside the human body, the spores swell, germinate, and progress to germ tube formation with hyphal extension. The hyphae invade the tissues and have a tendency to invade blood vessels where they cause thrombosis and tissue necrosis.

The immune system of healthy individuals helps to combat any kind of opportunistic infection through the innate system. Innate immune system with the alternative complement pathway, neutrophils, and phagocytic cells helps in destroying the spores of such infection. In patients with debilitating diseases such as hyperglycemia and acidosis, the innate immune system is able to destroy spores through the oxidative burst and no oxidative mechanism.

In addition, fungi require iron for growth and virulence. Conditions leading to high serum iron levels, such as diabetic ketoacidosis and COVID-19 (as seen in our patient) or hemochromatosis, predispose patients to mucormycosis. Acidosis likely disrupts the binding ability of iron-binding proteins, such as transferrin, allowing the fungi to utilize the free iron for growth. Individuals with high serum iron usually manifest higher chances of developing opportunistic infections such as mucormycosis.

The steroid therapy in COVID-19 patients suffering from diabetes is a bidirectional relationship. This relationship should be understood for better outcome as our patient was given steroid therapy and after that she developed opportunistic fungal infection.

The general health of this patient was good and she did not develop ketoacidosis; hence, the infection did not disseminate to other organs. Therefore, in this case, the patient had a localized rhino-maxillary form of the disease which is a subdivision of well-documented rhinocerebral mucormycosis. In the early stages of the disease, patients exhibit inflammation of facial spaces, diminished pain, facial cellulitis, paresthesia over the face, nasal discharge, necrotic turbinates, fever, headache, and lethargy.[4] However, in contrast to other reports, some of these symptoms were not present in this patient.

Imaging methods are little help during the early stages of rhinocerebral mucormycosis with thickening of the sinus mucosa or extraocular muscles being described as an early sign suggestive of the disease.[5] Computed tomography can show the involvement of the maxillofacial region correlating with the clinical finding, but not necessarily be accurate; however, magnetic resonance imaging usually has a more accurate picture of involvement and extent of the pathology both modalities can be evaluated simultaneously to have a complete involvement of the structures before planning of surgical interventions. Treatment of mucormycosis is carried out in three stages that are identification and compacting the underlying predisposing conditions, judicial advice of medication, and surgical interventions without much delay.[1],[4]

To summarize, this case report represents a variant of rhinocerebral mucormycosis case occurring in patient with previously identified predisposing factors. The characteristics and outcomes of such patients can be devastating if early diagnosis is not made. Therefore, early diagnosis of this opportunistic infection is of critical importance in COVID-19 patients with underlying conditions and those who have received steroid therapy because prompt diagnosis and aggressive treatment are essential for optimal outcomes.[6]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Jones AC, Bentsen TY, Freedman PD. Mucormycosis of the oral cavity. Oral Surg Oral Med Oral Pathol 1993;75:455-60.  Back to cited text no. 1
    
2.
Lehrer RI, Howard DH, Sypherd PS, Edwards JE, Segal GP, Winston DJ. Mucormycosis. Ann Intern Med 1980;93:93-108.  Back to cited text no. 2
    
3.
Eisenberg L, Wood T, Boles R. Mucormycosis. Laryngoscope 1977;87:347-56.  Back to cited text no. 3
    
4.
Abramson E, Wilson D, Arky RA. Rhinocerebral phycomycosis in association with diabetic ketoacidosis. Report of two cases and a review of clinical and experimental experience with amphotericin B therapy. Ann Intern Med 1967;66:735-42.  Back to cited text no. 4
    
5.
Rosenberg SW, Lepley JB. Mucormycosis in leukemia. Oral Surg Oral Med Oral Pathol 1982;54:26-32.  Back to cited text no. 5
    
6.
Tabarsi P, Khalili N, Pourabdollah M, Sharifynia S, Safavi Naeini A, Ghorbani J, et al. Case report: COVID-19-associated rhinosinusitis mucormycosis caused by Rhizopus arrhizus: A rare but potentially fatal infection occurring after treatment with corticosteroids. Am J Trop Med Hyg 2021;105:449-53.  Back to cited text no. 6
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]



 

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