Sinonasal mucormycosis with an unusual involvement of palate

Santosh Kumar Swain

doi:10.4103/am.am_130_21

CASE REPORT

Year : 2022 | Volume : 19 | Issue : 3 | Page : 177-179

Sinonasal mucormycosis with an unusual involvement of palate

Santosh Kumar Swain
Department of Otorhinolaryngology and Head and Neck Surgery, IMS and SUM Hospital, Siksha “O” Anusandhan University, Bhubaneswar, Odisha, India

Date of Submission	10-Nov-2021
Date of Decision	07-Dec-2021
Date of Acceptance	10-Dec-2021
Date of Web Publication	24-Jan-2022

Correspondence Address:
Santosh Kumar Swain
Department of Otorhinolaryngology and Head and Neck Surgery, IMS and SUM Hospital, Siksha “O” Anusandhan University, K8, Kalinga Nagar, Bhubaneswar - 751 003, Odisha
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/am.am_130_21

Abstract

Mucormycosis is caused by a fungal infection that belongs to the order of Mucorales and is one of the rapidly spreading infections of the human body. Rhizopus species are the most common etiological agent associated with mucormycosis. It is an emergency clinical condition and has a poor prognosis. The most common variant of the mucormycosis is rhinocerebral type which may spread to the orbit and brain. The identification of mucormycosis in the palatal region is often rare and late occurrence. A case of multiple ulcerations in the palatal region occurs due to sinonasal mucormycosis in a 55-year-old is reported. He was known case of diabetes mellitus and was diagnosed with coronavirus disease 2019 infection 1 year back but recovered with conservative treatment at home. Anterior rhinoscopy showed purulent discharge in both nostrils. He had no swelling in the face and neck. No tenderness was found in the head-and-neck region. He was successfully managed by surgical debridement combined with systemic liposomal amphotericin B for 6 weeks. The survival of patients is often dependent on early diagnosis and treatment.

Keywords: Amphotericin B, endoscopic debridement, mucormycosis, palatal ulcer

How to cite this article:
Swain SK. Sinonasal mucormycosis with an unusual involvement of palate. Apollo Med 2022;19:177-9

How to cite this URL:
Swain SK. Sinonasal mucormycosis with an unusual involvement of palate. Apollo Med [serial online] 2022 [cited 2023 Mar 23];19:177-9. Available from: https://apollomedicine.org/text.asp?2022/19/3/177/336568

Introduction

Mucormycosis is a deadly opportunistic infection caused by a saprophytic fungus from the Phycomycte family in the order Mucorales.^[1] Mucorales are saprophytes that can be found in decomposing organic materials and in the soil. These fungi are usually found on soil, manure, and fruits or vegetables.^[2] The nasal cavity and paranasal sinuses are often affected by fungal infection through inhaled dust.^[1] The common predisposing factors for mucormycosis are diabetic ketoacidosis and neutropenic conditions. This potentially fatal infection has a strong predilection for arteries. The fungi for mucormycosis are nonpathogenic but result in opportunistic serious infection in patients of diabetes mellitus, elderly age, under cytotoxic drugs, renal failure, leukemia, cirrhosis, and severe burns.^[2] Mucormycosis spread into orbit and brain leading to meningoencephalitis and cavernous sinus thrombosis which cause headache, facial swelling, orbital cellulitis, proptosis, loss of vision, ophthalmoplegia, necrosis of turbinates and palate as well as osteomyelitis of the bones in the facial area.^[3] The majority of the case reports of mucormycosis involving the facial area have been disseminated with orbital and intracranial involvement. In this report, we present a case of sinonasal mucormycosis that manifested as ulcers on the palate.

Case Report

A 55-year-old man presented to the otorhinolaryngology outpatient clinic with concerns of left nasal obstruction for 1 month and ulcers on the roof of the mouth for the past 15 days. There was mild pain on the left side of the face and a burning sensation in the roof of the oral cavity. He had a history of nasal discharge for 20 days from the left nostril. Around 15 days back, he developed ulcers over the palate, which rapidly increased in size and was interfering with swallowing food and interfering speech. He was known case of diabetes mellitus and was diagnosed with coronavirus disease 2019 (COVID-19) infection 1 year back but recovered with conservative treatment at home. During COVID-19 infection, he had mild symptoms of body ache, fever, mild cough, and no dyspnea. The medical history of the patient revealed that he was known case of uncontrolled Type 1 diabetes mellitus for the past 3 years and was on treatment (injection mixtard 20 units per day). The patient's father had Type 1 diabetes; therefore, there was a family history of diabetes. For the previous 10 years, the patient chewed tobacco. Oral cavity examination showed that three ulcers with 3 cm × 4 cm size each with irregular borders were appreciated over the palate [Figure 1]. Ulcers were covered by necrotic slough, part of the underlying bone was also exposed. Ulcers were nontender with few areas of erythema over the margins. Considering the medical history of the patient and rapidly proliferating ulcers over the palate, a provisional diagnosis of deep mycotic infection of the palate was made. The deadly midline granuloma and malignant ulcers were assumed to be differential diagnoses. Blood investigation showed increased erythrocyte sedimentation rate (35 mm/1^st h) and random blood sugar around 230 mg%. Computed tomography (CT) scan of the paranasal sinus showed mild sinusitis with focal erosion of the bony wall of the maxillary sinus. Tissue from palatal ulcer sent for histopathological examination. Numerous aseptate fungal hyphae branching at 90°, with neutrophilic infiltration entering the smaller blood vessels were seen in histological investigation (eosin staining) [Figure 2]. Magnetic resonance imaging (MRI) of the brain and orbit showed normal findings. The diagnosis of mucormycosis was confirmed. The patient underwent endoscopic debridement of the sinus and palatal necrotic tissue. He was administered Amphotericin B 1 mg/kg/day for 6 weeks.

Figure 1: Mucormycosis presenting with multiple palatal ulcers

Click here to view

Figure 2: Histopathological microphotograph showing nonseptate hyphae with 90° branching (Eosin stain, ×400)

Click here to view

A follow-up examination was done at 1 month, 3 months, and 6 months following completion of amphotericin injection which showed that nasal symptoms and oral cavity ulcerations resolved. Endoscopic examinations of the nasal cavity revealed normal nasal cavity, whereas oral cavity showed healed ulcerations at the palatal region at 3- and 6-month follow-up visits.

Discussion

Mucormycosis is a fulminating fungal infection that often involves the sinonasal area, oral cavity, and cranial and facial structures.^[4] It is an opportunistic fatal fungal infection that affects debilitating and immunosuppressive patients. Paltauf has first described mucormycosis in 1885.^[1] The causative fungi are ubiquitous, often harmless, and become pathogenic in human beings under certain conditions.^[5] Inhalation, ingestion, and percutaneous introduction of mucormycosis spores are the most common ways for this fungus to spread. The mucormycosis does not spread from person to person. The fungus has a strong liking for arteries and attaches to them. It spreads through the vessel's interior elastic lamina, causing thrombosis, ischemia, and necrosis of the surrounding tissues. Patients with mucormycosis often present with headaches, facial pain, malaise, swelling, and low-grade fever.^[6] In this case, the patient presented with nasal discharge, burning sensations in the mouth, and multiple ulcerations on the palatal area. There are usually four types of mucormycosis such as rhinocerebral, pulmonary, gastrointestinal, or disseminated. The rhinocerebral type is further classified into rhino-orbito-cerebral, which is invasive and can impact the ophthalmic and internal carotid arteries and rhinomaxillary, which affects the sphenopalatine and greater palatine arteries, causing turbinates thrombosis and palate necrosis.^[7] The differential diagnosis is midline lethal granuloma, Wegner's granulomatosis, necrotizing sialometaplasia, squamous cell carcinoma, and other deep-seated fungal infections.^[8] A malignant salivary gland neoplasm originating from the accessory glands of the palate can also be considered as a differential diagnosis for palatal involvement of the mucormycosis.^[9]

The timely diagnosis of the mucormycosis requires exact identification of the fungus with confirmation by culture.^[10] The culture, on the other hand, rarely evolves. Rhino-orbital-cerebral mucormycosis can be diagnosed by diagnostic nasal endoscopy and imaging. The spread of the infection in the sinonasal, brain, and skull base can be detected using a CT scan or MRI.^[11] Tissue biopsy is often confirmatory for establishing the diagnosis of mucormycosis.^[12] A severe form of mucormycosis is generally diagnosed clinically, and treatment should begin as soon as feasible before microbiological confirmation to provide the best possible clinical outcome.^[12] These fungi often thrive on common fungal culture media such as Sabouraud Dextrose Agar, inhibitory mold agar, or potato agar at temperatures ranging from 25°C to 55°C.^[13] Other microscopic procedures for direct Mucorales identification include 20% potassium hydroxide, Gomori's methenamine silver staining, periodic acid Schiff, and hematoxylin and eosin staining. Mucorales have hyphae that are ribbon like, wide, and aseptate, having right-angled hyphae.^[14] Histopathological pictures may show angioinvasion with the surrounding tissue. In this case, the histopathological study confirmed the diagnosis. By identifying fungus, polymerase chain reaction may be beneficial in determining the diagnosis of mucormycosis.^[15]

Despite early treatment, mucormycosis patients have a significant death rate, ranging from 16% to 100%.^[10] The mortality rate in rhinocerebral mucormycosis is approximately 67%.^[16] Mucormycosis advances quickly, necessitating immediate and intensive therapy. Aggressive and frequent surgical debridement of necrotic tissue, systemic antifungal medication, and prompt management of underlying systemic illnesses is all required for successful treatment.^[17]

Conclusion

A nonspecific ulcer overt the palate can be the presenting feature of mucormycosis. Therefore, clinicians need to be alert for early presentation of this disease, particularly during the evaluation of high-risk patients or immunocompromised patients. Early diagnosis and prompt treatment is a vital step for managing mucormycosis. Proper control of underlying systemic diseases associated with mucormycosis reduces the morbidity and mortality of this debilitating disease.

Informed consent

Patient consent was obtained.

Declaration of patient consent form

The authors certify that he has obtained all appropriate patient consent forms. In the form, the patient has given his consent for his 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 their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1.	Mignogna MD, Fortuna G, Leuci S, Adamo D, Ruoppo E, Siano M, et al. Mucormycosis in immunocompetent patients: A case-series of patients with maxillary sinus involvement and a critical review of the literature. Int J Infect Dis 2011;15:533-40.
2.	Auluck A. Maxillary necrosis by mucormycosis. A case report and literature review. Med Oral Patol Oral Cir Bucal 2007;12:E360-4.
3.	Kalaskar RR, Kalaskar AR, Ganvir S. Oral mucormycosis in an 18-month-old child: A rare case report with a literature review. J Korean Assoc Oral Maxillofac Surg 2016;42:105-10.
4.	Swain SK, Sahu MC, Banerjee A. Non-sinonasal isolated facio-orbital mucormycosis – A case report. J Mycol Med 2018;28:538-41.
5.	Reddy GC, Babu VR, Kumar MG, Rao VE. Mucormycosis: A case report. J Res Adv Dent 2015;4:29-32.
6.	Swain SK, Sahu MC, Baisakh MR. Mucormycosis of the head and neck. Apollo Med 2018;15:6-10. [Full text]
7.	Abidullah M, Kiran G, Gaddikeri K, Tanveer KG, Bhavirisetty D. Uncommon opportunistic fungal infections of oral cavity – Report of a case of rhino-orbital mucormycosis and review of literature. J Res Adv Dent 2015;4:51-5.
8.	Rai S, Misra D, Misra A, Jain A, Jain P, Dhawan A. Palatal mucormycosis masquerading as bacterial and fungal osteomyelitis: A rare case report. Contemp Clin Dent 2018;9:309-13. [PUBMED] [Full text]
9.	Desai V, Pratik P. Mucormycosis of oral mucosa: A rare case report. Int J Pharm Chem Boil Sci 2014;4:509-11.
10.	Swain SK, Jena PP, Das S, Gupta A. COVID-19 associated mucormycosis in head and neck region: Our experiences at a tertiary care teaching hospital of Eastern India. Siriraj Med J 2021;73:423-8.
11.	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.
12.	Spellberg B, Walsh TJ, Kontoyiannis DP, Edwards J Jr., Ibrahim AS. Recent advances in the management of mucormycosis: From bench to bedside. Clin Infect Dis 2009;48:1743-51.
13.	Swain SK, Behera IC, Mohanty JN. Mucormycosis in head-and-neck region – Our experiences at a tertiary care teaching hospital of Eastern India. Ann Indian Acad Otorhinolaryngol Head Neck Surg 2019;3:58-62. [Full text]
14.	Swain SK, Lenka S, Das SR. Rhino-orbital mucormycosis – A dreaded clinical entity. Int J Curr Res Rev 2020;12:197-203.
15.	Swain SK, Jena PP, Lenka S. COVID-19 associated mucormycosis in head and neck region of children during current pandemic: Our experiences. Pediatr Pol Pol J Paediatr 2021;96:162-7.
16.	Aggarwal P, Saxena S, Bansal V. Mucormycosis of maxillary sinus. J Oral Maxillofac Pathol 2007;11:66-9. [Full text]
17.	Mengji AK, Yaga US, Gollamudi N, Prakash B, Rajashekar E. Mucormycosis in a surgical defect masquerading as osteomyelitis: A case report and review of literature. Pan Afr Med J 2016;23:16.