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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 24  |  Issue : 1  |  Page : 60-62

Mucormycosis of middle ear in a diabetic patient


1 Nosocomial Infection Research Center, Isfahan University of Medical Sciences, Isfahan, Iran
2 Department of Otolaryngology, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran
3 Department of Infectious Diseases, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran
4 Department of Pathology, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran
5 Resident of Infectious Diseases, School of Medicine, Isfahan University of Medical Sciences, Isfahan, Iran

Date of Web Publication24-May-2018

Correspondence Address:
Dr. Atousa Hakamifard
Department of Infectious Diseases, School of Medicine, Isfahan University of Medical Sciences, Isfahan
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/indianjotol.INDIANJOTOL_85_17

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  Abstract 


Mucormycosis is an infection caused by fungi belonging to class zygomycetes, with high mortality and morbidity rate. Acquisition of mucormycosis is inhalation of spores or cutaneous route. The common risk factors for invasive mucormycosis consist of diabetes mellitus, high-dose glucocorticoid therapy, and neutropenia. The most clinical manifestation of mucormycosis is rhinocerebral lesions. Other manifestations are pulmonary, cutaneous, disseminated, and gastrointestinal. Ear involvement is extremely rare. The authors describe a case of mucormycosis cholesteatoma with concomitant central nervous system lesion in a patient with diabetes mellitus that responded to therapy.

Keywords: Cholesteatoma, diabetes mellitus, facial palsy, mucormycosis


How to cite this article:
Khorvash F, Reza Abtahi SH, Hakamifard A, Derakhshan M, Zarghami L. Mucormycosis of middle ear in a diabetic patient. Indian J Otol 2018;24:60-2

How to cite this URL:
Khorvash F, Reza Abtahi SH, Hakamifard A, Derakhshan M, Zarghami L. Mucormycosis of middle ear in a diabetic patient. Indian J Otol [serial online] 2018 [cited 2021 Apr 11];24:60-2. Available from: https://www.indianjotol.org/text.asp?2018/24/1/60/233135




  Introduction Top


Mucormycosis is an infection caused by fungi belonging to class zygomycetes, with high mortality and morbidity rate.[1],[2],[3] Zygomycoses are uncommon and frequently fatal diseases.[4] Agents of mucormycosis are in the environment including soil.[1],[5] Acquisition of mucormycosis is inhalation of spores or cutaneous route.[1] The common risk factors for invasive mucormycosis consist of diabetes mellitus, high-dose glucocorticoid therapy, and neutropenia.[6],[7],[8] Clinical manifestation of this invasive fungal infection is broad such as rhinocerebral, orbital, and pulmonary.[1] Fungal infections of the central nervous system, particularly cerebral mucormycosis or brain abscess are very rare.[9] Brain abscess due to mucormycosis, particularly present in intravenous drug abusers.[1] Mucormycosis is seen more commonly in the nose and paranasal sinuses.[10] Mucor cholesteatoma is an extremely rare entity. The authors describe a case of a diabetic patient with cholesteatoma associated with mucormycosis and concomitant brain abscess.


  Case Report Top


A 64-year-old, diabetic male was admitted to Alzahra Hospital with a history of sudden right ear pain and right ear hearing loss which accompanied by mucopurulent otorrhea and facial asymmetry. He also gave a history of an occasional headache 1 month before, which resolved with analgesic drugs. The patient had a history of diabetes mellitus. On admission, physical examination revealed facial nerve palsy. His right ear hearing was significantly reduced when compared to the other side. Laboratory study showed white blood cell = 5100, Hb = 9.4, PLT = 170,000. Serum creatinine was 1.4 mg/dl. Level of serum erythrocyte sedimentation rate was 67. After evaluation, cholesteatoma was confirmed, and mastoidectomy and fistula repair were done. Antrum was full of cholesteatoma. Furthermore, tympanoplasty was performed. Brain magnetic resonance imaging (MRI) revealed single ring-enhancing lesion with peripheral edema in right temporal lobe consistent with brain abscess [Figure 1] and [Figure 2]. An empiric antimicrobial regimen including ceftriaxone and metronidazole were started. Histopathology of the specimen revealed mucormycosis, an unusual presentation of mucor in the middle ear [Figure 3]. The findings were compatible with the diagnosis of middle ear mucormycosis and concomitant temporal lobe brain abscess. Hence, the administration of ceftriaxone and metronidazole were stopped, and amphotericin was started. The liposomal amphotericin B, 5 mg/kg was started along with diabetic management. Control brain MRI was done after 2 weeks which revealed improvement of cerebral lesion.
Figure 1: Ring enhancement lesion in temporal lobe

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Figure 2: Ring enhancement lesion in temporal lobe compatible with brain abscess

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Figure 3: Histopathology of the specimen revealed broad and aseptate hyphae, compatible with mucormycosis

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  Discussion Top


In this case report, a patient with middle ear mucormycosis and the concomitant cerebral lesion was described. Mucormycosis is a rare infection caused by fungi belonging to class zygomycetes.[11] Factors associated with an increased risk of mucormycosis include immunosuppression, diabetes mellitus, and administration of high dose corticosteroids.[1] The most clinical manifestation of mucormycosis is rhinocerebral lesions. Other manifestations are pulmonary, cutaneous, disseminated, and gastrointestinal.[12] Ear involvement is extremely rare.[10],[13] The patient was a controlled diabetic patient. Uncontrol diabetes has been introduced as a risk factor for this invasive fungal infection.[12] Tissue histology is still the most important diagnostic approach for mucormycosis. Histopathological study of specimens obtained from a biopsy of middle ear was leading to the diagnosis of mucormycosis. The cholesteatoma was associated with perineural involvement and causes facial palsy. In this patient, intravenous antifungal therapy without surgical intervention for brain abscess resulted in significant clinical improvement. We believe that if the fungal abscess were secondarily infected with bacteria, the lesions would relapse or would not resolve primarily with pure antifungal treatment. The involvement of the middle ear in mucormycosis with facial paralysis is extremely rare. Studies show very few cases of temporal bone mucormycosis that have been reported, in diabetic and nondiabetic people.[10],[14],[15]


  Conclusion Top


Physicians should be aware of such atypical clinical presentation of this fungal infection. This would facilitate implementation of an early appropriate medical and surgical treatment. Such a treatment regimen would aid better outcome.

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.
Kontoyiannis DP, Lewis RE. Agent of mucormycosis and entomophtoramycosis. Mandell, Douglas, and Bennett's Principles and Practice of Infectious Diseases. 8th ed. Philadelphia: Churchill Livingstone; 2015. p. 2909-19.  Back to cited text no. 1
    
2.
Ibrahim AS, Spellberg B, Walsh TJ, Kontoyiannis DP. Pathogenesis of mucormycosis. Clin Infect Dis 2012;54 Suppl 1:S16-22.  Back to cited text no. 2
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3.
Walsh TJ, Skiada A, Cornely OA, Roilides E, Ibrahim A, Zaoutis T, et al. Development of new strategies for early diagnosis of mucormycosis from bench to bedside. Mycoses 2014;57 Suppl 3:2-7.  Back to cited text no. 3
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4.
Mohamed MS, Abdel-Motaleb HY, Mobarak FA. Management of rhino-orbital mucormycosis. Saudi Med J 2015;36:865-8.  Back to cited text no. 4
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5.
Ananthaneni AR, Undavalli SB, Velagapudi RP, Guduru VS. Mucormycosis: An atrocious mate of patients with diabetes. BMJ Case Rep 2013;2013. pii: bcr2013009600.  Back to cited text no. 5
    
6.
Vijayabala GS, Annigeri RG, Sudarshan R. Mucormycosis in a diabetic ketoacidosis patient. Asian Pac J Trop Biomed 2013;3:830-3.  Back to cited text no. 6
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7.
Spellberg B, Kontoyiannis DP, Fredricks D, Morris MI, Perfect JR, Chin-Hong PV, et al. Risk factors for mortality in patients with mucormycosis. Med Mycol 2012;50:611-8.  Back to cited text no. 7
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8.
Teixeira CA, Medeiros PB, Leushner P, Almeida F. Rhinocerebral mucormycosis: Literature review apropos of a rare entity. BMJ Case Rep 2013;2013. pii: bcr2012008552.  Back to cited text no. 8
    
9.
Ma J, Jia R, Li J, Liu Y, Li Y, Lin P, et al. Retrospective clinical study of eighty-one cases of intracranial mucormycosis. J Glob Infect Dis 2015;7:143-50.  Back to cited text no. 9
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10.
Hazarika P, Zachariah J, Victor J, John M, Devi C, Abraham P, et al. Mucormycosis of the middle ear: A case report with review of literature. Indian J Otolaryngol Head Neck Surg 2012;64:90-4.  Back to cited text no. 10
    
11.
Singh ID, Galagali JR, Kumar S. Otocerebral mucormycosis: A rare case in a diabetic patient with unusual presentation. J Otolaryngol ENT Res 2015;2:00020.  Back to cited text no. 11
    
12.
Petrikkos G, Skiada A, Lortholary O, Roilides E, Walsh TJ, Kontoyiannis DP, et al. Epidemiology and clinical manifestations of mucormycosis. Clin Infect Dis 2012;54 Suppl 1:S23-34.  Back to cited text no. 12
    
13.
Kermani W, Bouttay R, Belcadhi M, Zaghouani H, Ben Ali M, Abdelkéfi M, et al. ENT mucormycosis. Report of 4 cases. Eur Ann Otorhinolaryngol Head Neck Dis 2016;133:83-6.  Back to cited text no. 13
    
14.
Yun MW, Lui CC, Chen WJ. Facial paralysis secondary to tympanic mucormycosis: Case report. Am J Otol 1994;15:413-4.  Back to cited text no. 14
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15.
Dias FV, Goretti BB, Ramirez EP, dos Reis JC, da Silva Almeida J, Guerreiro SM. Cholesteatoma of the middle ear associated with mucormycosis: A case report. Int Arch Otorhinolaryngol 2014;18:2365.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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