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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 14  |  Issue : 4  |  Page : 397-400

Mucormycosis of maxillary sinus in a newly diagnosed case of diabetes mellitus


Department of Oral and Maxillofacial Surgery, Sharad Pawar Dental College, Datta Meghe Institute of Medical Sciences, Wardha, Maharashtra, India

Date of Submission31-Oct-2019
Date of Decision05-Nov-2019
Date of Acceptance30-Nov-2019
Date of Web Publication16-Jul-2020

Correspondence Address:
Dr. Pawan Hingnikar
Department of Oral and Maxillofacial Surgery, Sharad Pawar Dental College, Datta Meghe Institute of Medical Sciences, Wardha, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdmimsu.jdmimsu_170_19

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  Abstract 


Rhinocerebral mucormycosis is the third most common fungal infection after aspergillosis and candidiasis. The importance of mucormycosis has grown in recent years, as the number of patients has increased dramatically. Herein, we present a case of a newly diagnosed patient of diabetes mellitus having ulcerated palate and destruction of maxillary sinus evident on computed tomography scan. A biopsy report was suggestive of mucormycosis. Aggressive medical and surgical treatment was done along with the treatment of diabetes mellitus.

Keywords: Diabetes mellitus, mucormycosis, rhinocerebral mucormycosis


How to cite this article:
Hingnikar P, Bhola N, Jadhav A, Sharma A. Mucormycosis of maxillary sinus in a newly diagnosed case of diabetes mellitus. J Datta Meghe Inst Med Sci Univ 2019;14:397-400

How to cite this URL:
Hingnikar P, Bhola N, Jadhav A, Sharma A. Mucormycosis of maxillary sinus in a newly diagnosed case of diabetes mellitus. J Datta Meghe Inst Med Sci Univ [serial online] 2019 [cited 2020 Aug 11];14:397-400. Available from: http://www.journaldmims.com/text.asp?2019/14/4/397/289840




  Introduction Top


Mucormycosis, also known as zygomycosis or phycomycosis, is an acute invasive fungal infection characterized by necrotizing soft and hard tissues. It is the most common invasive fungal infection after aspergillosis and candidiasis.[1] Variations of mucormycosis include, rhinocerebral, pulmonary, gastrointestinal, cutaneous, and disseminated infections. Risk factors for such infections include diabetes, ketoacidosis, lymphoma, leukemia, neutropenia, corticosteroid use, long-term immunosuppressive therapy, or deferoxamine therapy.[2] In developed countries, the disease remains uncommon, and at present, it is mostly seen in patients with diabetes mellitus and hematological malignancies (HMs) undergoing chemotherapy and in those who have received allogeneic stem cell transplants.[3] In contrast, in developing countries, especially in India, mucormycosis cases, although sporadic, occur mainly in patients with uncontrolled diabetes or trauma.[4] The mortality rates range from 16% in cutaneous mycosis up to 100% in gastrointestinal or disseminated infection with an overall mortality of 36%–44%. Rhinocerebral mucormycosis occurs in about 39% of the cases of mucormycosis, with an associated mortality range of approximately 62%.[1]


  Case Report Top


A 44-year-old female patient presented to the Department of Oral and Maxillofacial Surgery, Sharad Pawar Dental College, Sawangi, Wardha, with a history of painful nonhealing ulcer of size approximately 2 cm × 2 cm in the palate for 1 month. One month back, she underwent extraction of 12 11 21 22 in a private clinic where she was diagnosed with diabetes mellitus during routine hematological examination for which she is under medications (tablet Gudpress AM 50 and tablet Afoglip). The patient was referred to a private hospital for the management of ulcerative lesion over the palate, where she underwent incisional biopsy, report suggestive of “Fungal infection of maxillary sinus – Mucormycosis.”

On examination, the patient had a diffused swelling over the left cheek region which was soft in consistency and tender in nature. Oral cavity examination showed yellowish discoloration of the anterior palatal region and left alveolar ridge [Figure 1]. There was no significant evidence of cervical lymphadenopathy.
Figure 1: Clinical presentation of palatal ulceration with necrotic bone

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Contrast-enhanced computed tomography (CECT) was done [Figure 2]. CECT revealed the destruction of the anterior, medial, and posterolateral wall of the left maxillary sinus with an underlying mucosal thickening. Anterior wall destruction extended to the adjacent nasal bone.
Figure 2: Axial section showing destruction of the anterior, medial, and posterolateral wall of the left maxillary sinus, extending into the adjacent nasal bone

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Medical intervention

The patient was given liposomal amphotericin 3 mg/kg IV for 3 weeks. During this period, regular kidney function tests were carried out as it has a nephrotoxic effect.

Surgical intervention

Prior to surgical intervention, the patient was kept on injection insulin in a sliding scale. The patient then underwent surgical debridement [Figure 3] under general anesthesia where all the necrotic tissues of the palate and maxilla were debrided and closure done with 3-0 vicryl suture. The patient received piperacillin–tazobactam and metronidazole for 7 days. After the surgery, the patient was again given liposomal amphotericin B for 2 weeks.
Figure 3: Surgical debridement of necrotic tissue

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Follow-up condition

At 2 years of follow-up, the surgical site healed satisfactorily; however, a fistula of size 2 mm × 2 mm developed in the anterior region of the palate for which the patient was advised for obturator [Figure 4].
Figure 4: Postoperative intraoral view showing a healed surgical site

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  Discussion Top


Mucormycosis is a type of infection caused by a fungus belonging to the order Mucorales. It was first described by Paulltauf A in 1885.[5] Clinically, most commonly recovered Mucorale species belong to the genera Rhizopus, Lichtheimia, and Mucor. Other less commonly found genera are Rhizomucor, Saksenaea, Cunninghamella, and Apophysomyces. The incidence of rhinocerebral mucormycosis in India is 0.14 cases/1000 population.[6]

Mucormycosis is a formidable threat in people with diabetes. In India, a study performed in a tertiary care center showed that 74% of patients with mucormycosis had uncontrolled diabetes, whereas 43% of these cases were diagnosed for the first time for diabetes,[7] as seen in our case.

Mucorales invade deep tissues via percutaneous inoculation, inhalation of airborne spores, or ingestion.[8] After exposure to the deep tissues, it begins to grow and invades the arterial wall. This causes arteritis which, in turn, leads to the formation of vascular thrombus. Vascular thrombosis eventually leads to tissue necrosis. In diabetic patients, Mucorale species cause the production of enzyme ketoreductase. This leads to a state of hyperglycemia which favors the fungal growth.

The diagnosis is based on the evidence of soft- and hard-tissue necrosis. A definitive diagnosis can only be made after histopathological examination. Typical nonseptated hyphae indicate the involvement of Mucorale species. These hyphae typically invade the blood vessel walls and cause infarction. This results in necrotic areas beneath highly inflamed tissue. CT scan provides a clear image of the hard and soft tissues involved.

Clinically, rhinocerebral mucormycosis may manifest as necrosis of the palate or sinuses, thus more appropriately termed as rhinosinus mucormycosis when cerebral content not involved. Black sores present on the palate or nasal mucosa may suggest mucormycosis in the appropriate clinical context. The most frequent presenting symptoms are similar to that of bacterial rhinosinusitis, such as fever, unilateral facial swelling with pain, or orbital cellulitis. Cavernous sinus thrombosis and cranial invasion may be the sequelae of untreated rhinosinus mucormycosis.[9]

Early treatment after an accurate diagnosis is crucial for achieving a better prognosis in treating mucormycosis. Treatment consists of systemic antifungal treatment, causative therapy for the underlying disease, and radical surgical debridement. Conventional antifungal treatment involves injection amphotericin B (dose 1–1.5 mg/kg/day) and its lipid formulation (3–5 mg/kg/day). Lipid formulations of amphotericin B are considered superior over amphotericin B deoxycholate in the treatment of mucormycosis, as the former has less nephrotoxic effects, better penetration to the brain, and has superior immunomodulatory effects.[10]

The European Society of Clinical Microbiology and Infectious Diseases and European Confederation of Medical Mycology recommended immediate first-line antifungal therapy followed by radical surgical debridement. Since thrombosis and tissue necrosis associated with mucormycosis lead to poor penetration of antifungal agents at the sites of infection and compromise their efficacy, surgical debridement must be performed very aggressively. Most debridements may cause large defects which may require rehabilitation by hard- and soft-tissue reconstruction.

Apart from treating the underlying causes such as metabolic diseases, other options include leukocyte transfusions, treatment with interferon-gamma, hyperbaric oxygenation, iron-chelating agents, and colistin.[10],[11],[12],[13],[14],[15]

In conclusion, an early diagnosis with aggressive medical and surgical treatment and correction of any immunosuppressing factors are considered the fundamental pillars in a successful outcome. When intracranial involvement occurs, the outcome is almost fatal.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/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, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Metzen D, Böhm H, Zimmermann M, Reuther T, Kübler AC, Müller-Richter UD. Mucormycosis of the head and neck. J Craniomaxillofac Surg 2012;40:e321-7.  Back to cited text no. 1
    
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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.  Back to cited text no. 2
    
3.
Chakrabarti A, Das A, Sharma A, Panda N, Das S, Gupta KL, et al. Ten years' experience in zygomycosis at a tertiary care centre in India. J Infect 2001;42:261-6.  Back to cited text no. 3
    
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Spellberg B, Edwards J Jr., Ibrahim A. Novel perspectives on mucormycosis: Pathophysiology, presentation, and management. Clin Microbiol Rev 2005;18:556-69.  Back to cited text no. 4
    
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Mallis A, Mastronikolis SN, Naxakis SS, Papadas AT. Rhinocerebral mucormycosis: An update. Eur Rev Med Pharmacol Sci 2010;14:987-92.  Back to cited text no. 5
    
6.
Chakrabarti A, Singh R. Mucormycosis in India: Unique features. Mycoses 2014;57 Suppl 3:85-90.  Back to cited text no. 6
    
7.
Chakrabarti A, Das A, Mandal J, Shivaprakash MR, George VK, Tarai B, et al. The rising trend of invasive zygomycosis in patients with uncontrolled diabetes mellitus. Med Mycol 2006;44:335-42.  Back to cited text no. 7
    
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Prabhu RM, Patel R. Mucormycosis and entomophthoramycosis: A review of the clinical manifestations, diagnosis and treatment. Clin Microbiol Infect 2004;10 Suppl 1:31-47.  Back to cited text no. 8
    
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González Ballester D, González-García R, Moreno García C, Ruiz-Laza L, Monje Gil F. Mucormycosis of the head and neck: Report of five cases with different presentations. J Craniomaxillofac Surg 2012;40:584-91.  Back to cited text no. 9
    
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Sun HY, Singh N. Mucormycosis: Its contemporary face and management strategies. Lancet Infect Dis 2011;11:301-11.  Back to cited text no. 10
    
11.
Bhinder HHPS, Kamble TK. The Study of Carotid Intima-Media Thickness in Prediabetes and Its Correlation with Cardiovascular Risk Factors. J Datta Meghe Inst Med Sci Univ 2018;13:79-82. Available from: https://doi.org/10.4103/jdmimsu.jdmimsu_58_18. [Last accessed on 2019 Oct 17].  Back to cited text no. 11
    
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Rathi N, Taksande B, Kumar S. Nerve Conduction Studies of Peripheral Motor and Sensory Nerves in the Subjects with Prediabetes. J Endocrinol Metab 9, no. 5 (2019): 147–50. Available from: https://doi.org/10.14740/jem602. [Last accessed on 2019 Oct 17].  Back to cited text no. 12
    
13.
Walinjkar RS, Khadse S, Kumar S, Bawankule S, Acharya S. Platelet Indices as a Predictor of Microvascular Complications in Type 2 Diabetes. Indian J Endocrinol Metab 2019;23:206-10. Available from: https://doi.org/10.4103/ijem.IJEM-13-19. [Last accessed on 2019 Oct 17].  Back to cited text no. 13
    
14.
Cladius S, Jadhav U, Ghewade B, Ali S, Dhamgaye T. Study of Diabetes Mellitus in Association with Tuberculosis. J Datta Meghe Inst Med Sci Univ 2017;12:143-7. Available from: https://doi.org/10.4103/jdmimsu.jdmimsu_62_17. [Last accessed on 2019 Oct 17].  Back to cited text no. 14
    
15.
Singh P, Jain S, Methwani D, Kalambe S, Chandravanshi D, Gaurkar S, et al. Study of Correlation of Pre-Operative Findings with Intra-Operative Ossicular Status in Patients with Chronic Otitis Media. Iran J Otorhinolaryngol 2018;30:273-81. Available from: https://doi.org/10.22038/ijorl.2018.26795.1875. [Last accessed on 2019 Oct 17].  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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