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CASE REPORT
Year : 2019  |  Volume : 46  |  Issue : 3  |  Page : 106-109

Invasive aspergillosis of tympanomastoid cavity


1 Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha “O” Anusandhan University, Bhubaneswar, Odisha, India
2 Department of Oral Pathology and Microbiology, Institute of Dental Sciences, Siksha “O” Anusandhan University, Bhubaneswar, Odisha, India
3 Central Research Laboratory, IMS and SUM Hospital, Siksha “O” Anusandhan University, Bhubaneswar, Odisha, India

Date of Web Publication28-Jan-2020

Correspondence Address:
Dr. Santosh Kumar Swain
Department of Otorhinolaryngology, IMS and SUM Hospital, Kalinga Nagar, Bhubaneswar - 751 003, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jss.JSS_41_19

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  Abstract 


Invasive aspergillosis of the tympanomastoid cavity is an extremely rare clinical incidence in comparison to the invasive fungal infections of the paranasal sinus and other parts of the body. Delay in the diagnosis and treatment of this rare clinical entity often leads to fatal complications such as skull base osteomyelitis and progressive multiple cranial nerve palsies. The invasive aspergillosis of the tympanomastoid region should be considered as a differential diagnosis for atypical mastoiditis in immunocompromised patients. Here, we report a case of invasive aspergillosis of the tympanomastoid region in a 60-year-old diabetic patient with facial nerve palsy. The diagnosis was done by the demonstration of fungi in the tissue with Gomori methenamine silver staining and confirmed with molecular diagnosis. Surgical debridement of the tympanomastoid region was done along with long-term voriconazole therapy.

Keywords: Invasive aspergillosis, surgical debridement, tympanomastoid cavity, voriconazole


How to cite this article:
Swain SK, Debta P, Bishoyi AK, Padhy R. Invasive aspergillosis of tympanomastoid cavity. J Sci Soc 2019;46:106-9

How to cite this URL:
Swain SK, Debta P, Bishoyi AK, Padhy R. Invasive aspergillosis of tympanomastoid cavity. J Sci Soc [serial online] 2019 [cited 2020 Feb 24];46:106-9. Available from: http://www.jscisociety.com/text.asp?2019/46/3/106/277002




  Introduction Top


Invasive respiratory aspergillosis is often found in an extremely immunocompromised host, whereas the segregation of Aspergillus species in the immunocompetent host may be due to colonization.[1],[2] Tympanomastoid cavity includes the middle ear and mastoid air cells which are parts of the temporal bone. Fungal infection of this region is rarely observed in routine clinical practice. Isolated invasive aspergillosis of the tympanomastoid cavity is a rare and potential life-threatening clinical entity. Aspergillus is a ubiquitous saprophytic mold, seen often on the decaying material. Inhalations of the spores occur through nose which causes infections of the paranasal sinuses and other sites of the body. Invasive aspergillosis of the tympanomastoid cavity is extremely rare although Aspergillus species are often implicated in otomycosis. The first case of invasive aspergillosis of the tympanomastoid region of the temporal bone was documented in medical literature in 1985 on a patient of acute myeloid leukemia.[3] The diagnosis of this lesion is still remains a diagnostic challenge for a clinician. It requires a high degree of suspicion to reach a diagnosis, so as to start early treatment and prevent its potentially lethal complications. Here, we report a case of invasive aspergillosis of the tympanomastoid region of the temporal bone with facial palsy in an elderly diabetic patient.


  Case Report Top


A 60-year-old male attended the outpatient department of otorhinolaryngology for the right ear discharge for 2 years and ear pain for 1 month. He was a known case of diabetic since of 10 years, on irregular treatment. On examination, there was grayish mass in the external auditory canal with total perforation of tympanic membrane. There was right side facial weakness for 1 month. Other examinations of ear, nose, and throat were within the normal limits. Pure tone audiogram demonstrated a severe mixed hearing loss on the right side with normal hearing level on the left side. Facial nerve examination showed the right side lower motor facial nerve palsy of House–Brackmann Grade III. Laboratory report revealed raised fasting blood sugar (350 mg/dl), postprandial blood sugar (420 mg/dl), and raised erythrocyte sedimentation rate (80 mm/h). There were no ketone bodies found in the urine. The discharge from the external auditory canal sent for culture sensitivity which showed no bacterial growth. The patient was started with broad-spectrum antibiotic for anticipating unsafe chronic otitis media or skull base osteomyelitis along with appropriate dose of insulin for controlling diabetes. For assessing the extent of disease and facial nerve status, high-resolution computed tomography (CT) was done. CT scan of the temporal bone showed soft-tissue mass filling the right external ear canal, middle ear, and mastoid cavity with erosion of the vertical segment of the facial nerve [Figure 1]. The patient was planned for the right mastoid exploration and facial nerve decompression. Intraoperative findings revealed a mass arising from the posterior ear canal and eroding the vertical canal of the facial nerve. The mass was extending into the mastoid antrum, aditus, and middle ear [Figure 2]. Ossicles in the middle ear were absent. There was no cholesteatoma seen in the mastoid cavity, attic, and middle ear. The mass from the tympanomastoid cavity was cleaned and facial nerve decompression done, and the mass was sent for histopathological examination and fungal culture. The wound was closed with large meatoplasty for proper ventilation and drainage of the mastoid cavity. The histopathological examination of the mass removed from the tympanomastoid cavity showed nonspecific inflammation along with granulomatous tissue and presence of lymphocytes, plasma cells, neutrophils, and eosinophils. Culture and staining for tuberculosis were negative. Culture of the tissue from tympanomastoid cavity on Sabouraud's dextrose agar revealed yellowish-green granular mold with radially arranged conidia suggest Aspergillus flavus [Figure 3]a. Under microscopic image with help of cotton blue staining, it revealed A. flavus [Figure 3]b. Gomori methenamine silver (GMS) staining of the biopsy material showed acutely branching hyphae confirming the A. flavus [Figure 4]. For more conformation, we went through the molecular study. With polymerase chain reaction, using universal fungal primers forward, 5'- TCC GTA GGT GAA CCT GCG G- 3'. and reverse, 5'- TTG GTC CGT GTT TCA AGA CG- 3.' generated the 28S rRNA region amplicons for sequencing. The sequence was analyzed with BLAST online software (Sequencing Analysis Software v6.0), and it was revealed A. flavus.
Figure 1: Computed tomography scan of the right side temporal bone (axial cut) showing pathology in the mastoid antrum and middle ear (arrow mark)

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Figure 2: Intraoperative picture showing fungal material filling the mastoid antrum (arrow mark)

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Figure 3: (a) Macroscopic view of 48 h culture of Aspergillus flavus, (b) microscopic view of the vesicles and hyphal forms of Aspergillus flavus (×400)

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Figure 4: Microscopic image of Aspergillus flavus with Gomori methanamine silver staining (×400)

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The patient was started with an antifungal drug such as voriconazole. After treatment with tablet voriconazole 200 mg twice daily for 2 months, the lesions have resolved as per clinical examinations. The facial paralysis was also improved to normal clinically (House–Brackmann Grade I) after 2 weeks of surgery. Pure tone audiogram [Figure 5] showed same hearing loss after 2 months of surgery as before. The patient was under regular follow-up. The patient details are given in [Table 1].
Figure 5: Pure tone audiogram showing the right side moderate mixed hearing loss

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Table 1: Patient details including the clinical presentations, diagnosis, treatment, and follow-up

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  Discussion Top


Invasive aspergillosis is a common opportunistic fungal infection among immunocompromised patients. Aspergillus is a saprophytic fungus seen worldwide. The most common isolate of Aspergillus is Aspergillus fumigatus. Fungal infections of the tympanomastoid region are often reported due to the infections of A. fumigatus in immunocompromised patient, whereas few cases are also seen in immunocompetent hosts.[4] The risk factors for invasive fungal infections are cytotoxic drugs, prolonged neutropenia, graft versus host disease, irradiation, systemic corticosteroids, and human immunodeficiency virus infections.[5] The invasive fungal infections of the tympanomastoid region is a rare incidence in comparison to the invasive fungal sinusitis, as there is barrier of the eardrum and the absence of exposure of the  Eustachian tube More Details to inhaled fungal spores. The maxillary sinus and lungs are the most common locations for primary aspergillosis.[6] Fungal infection of the external auditory canal or otomycosis is a common disease of the ear without any morbidity and mortality. The fungal infections of the middle ear and mastoid are extremely rare incidence in clinical practice. It may be seen in immunocompromised patients such as those of acquired immunodeficiency syndrome, leukemia, uncontrolled diabetes mellitus, and patients under immunosuppressive drugs. Very rarely, it is seen in immunocompetent persons.[7] The temporal bone involvement of aspergillosis is classified into three forms: noninvasive type, invasive type, and fulminant type. Invasive type of aspergillosis causes temporal bone erosion and has granulomatous response. The fulminant type is often seen in immunocompromised patients, and the characteristic features are angioinvasion, rapid progression, and minimal tissue reaction.[8]

The clinical presentations of invasive aspergillosis of the tympanomastoid cavity are otorrhea which does not responds to medical treatment and hearing loss. Facial nerve paralysis may occur with erosion of facial nerve canal. Facial nerve palsy with chronic otitis media in immunocompromised patients will give rise to suspicion of invasive aspergillosis of tympanomastoiditis.[9] Invasive aspergillosis is often found among patients with lymphoproliferative disorders, but it may be seen among patients with variety of the diseases of humoral or cell-mediated immunity. The successful management of this disease requires early diagnosis and treatment. The early diagnosis needs a high index of suspicion of the susceptible individuals, including those associated with immunocompromised state. The presence of fungi on culture media alone is not conclusive of invasive fungal infection of the tympanomastoid region. Demonstrations of the fungus in the tissue, as in our case, with GMS staining suggest the invasive nature of the fungal infection. CT scan of the temporal bone is useful for showing bony erosion by aspergillosis. The facial canal erosion, labyrinthine erosion, and erosion of the tegmen are easily recognized in the axial and coronal cuts of the CT scan of the temporal bone. Intracranial involvement or soft-tissue involvement are better delineated by the magnetic resonance imaging which appears as hypointense signals on T1-weighted and T2-weighted images and as heterogeneous and multiple peripheral enhancements on postgadolinium T1-weighted images.[10] If the diagnosis is confirmed, treatment of the invasive aspergillosis includes antifungal treatment, surgical debridement, and controlling of the underlying immunodeficiency conditions. In refractory cases, hyperbaric oxygen therapy may be considered although its effectiveness is still doubtful.[11] Aggressive surgical debridement is treatment of choice in most of the cases. In our case, surgical debridement of the tympanomastoid region was done in the affected side. The antifungal agent of choice is amphotericin-B, but due to its severe systemic side effects losing its favor. Itraconazole has its good outcome either alone or along with amphotericin-B.[12] Voriconazole is a newer generation azole group of antifungal agent. It is more effective for Aspergillus sp., in comparison to other azoles and amphotericin-B except more cost.[13] Voriconazole acts by blocking the synthesis of ergosterol through the inhibition of CYP-dependent lanosterol 14α-demethylase. The treatment period of antifungal therapy varies with duration ranging from 3 to 77 weeks.[14] In this case, voriconazole was given with successful outcome. Recently, isavuconazole which is a triazole antifungal agent, approved as the treatment option for invasive fungal infections, such as aspergillosis.[15] The prognosis of the invasive aspergillosis is closely related to the site of the involvement, extension of the lesion, and immunity status of the patient. Early diagnosis of the disease on the basis of the radiology and pathological findings and with treatment options such as surgical debridement and antifungal treatment possibly enhance the prognosis of the patient.


  Conclusion Top


The diagnosis of invasive aspergillosis of the tympanomastoid region needs a high index of suspicion, as it is often misdiagnosed as unsafe type of chronic otitis media or malignant otitis externa. It is often associated with a neutropenic state of the patient or immunocompromised state. Antifungal treatment with aggressive debridement of the disease and restitution of the immunocompetent state are important part of the patient management. Without proper intervention at early stage, mortality and morbidity may happen in relation to facial nerve palsy, hearing loss, vertigo, and even death.

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.
Crum-Cianflone NF. Invasive aspergillosis associated with severe influenza infections. Open Forum Infect Dis 2016;3:1-8.  Back to cited text no. 1
    
2.
Patterson TF, Thompson GR 3rd, Denning DW, Fishman JA, Hadley S, Herbrecht R, et al. Practice Guidelines for the Diagnosis and Management of Aspergillosis: 2016 Update by the Infectious Diseases Society of America. Clin Infect Dis 2016;63:e1-e60.  Back to cited text no. 2
    
3.
Petrak R, Pottage J, Levin S. Invasive external otitis caused by Aspergillus fumigatus in an immunocompromised patient. J Infect Dis 1985;51:196.  Back to cited text no. 3
    
4.
Mani R, Belcadhi M, Ben Said M, Bouzouita K. Aspergillus mastoiditis with intact tympanic membrane in immunocompetent host. B-ENT 2008;4:39-43.  Back to cited text no. 4
    
5.
Aycicek A, Kenar F, Demirdal T, Asik G, Haktanir A, Sargin R, et al. Facial paralysis due to invasive Aspergillus of the temporal bone in an immunocompetent child. Int J Pediatr Otorhinolaryngol Extra 2009;4:143-6.  Back to cited text no. 5
    
6.
Shah S, Shirani P, Schmolck H, Young WC, Schulz PE. CNS aspergillosis in a patient with Crohn's disease on immunosuppressants: A case report. Cases J 2009;2:6376.  Back to cited text no. 6
    
7.
van Tol A, van Rijswijk J. Aspergillus mastoiditis, presenting with unexplained progressive otalgia, in an immunocompetent (older) patient. Eur Arch Otorhinolaryngol 2009;266:1655-7.  Back to cited text no. 7
    
8.
Bradoo RA, Shah KD, Gayathri H, Kapadia MA. Invasive aspergillosis of the temporal bone. Indian J Otol 2012;18:30-3.  Back to cited text no. 8
  [Full text]  
9.
Lyos AT, Malpica A, Estrada R, Katz CD, Jenkins HA. Invasive aspergillosis of the temporal bone: An unusual manifestation of acquired immunodeficiency syndrome. Am J Otolaryngol 1993;14:444-8.  Back to cited text no. 9
    
10.
Azarpira N, Esfandiari M, Bagheri MH, Rakei S, Salari S. Cerebral aspergillosis presenting as a mass lesion. Braz J Infect Dis 2008;12:349-51.  Back to cited text no. 10
    
11.
Ling SS, Sader C. Fungal malignant otitis externa treated with hyperbaric oxygen. Int J Infect Dis 2008;12:550-2.  Back to cited text no. 11
    
12.
Denning DW, Tucker RM, Hanson LH, Stevens DA. Treatment of invasive aspergillosis with itraconazole. Am J Med 1989;86:791-800.  Back to cited text no. 12
    
13.
Herbrecht R, Denning DW, Patterson TF, Bennett JE, Greene RE, Oestmann JW, et al. Voriconazole versus amphotericin B for primary therapy of invasive aspergillosis. N Engl J Med 2002;347:408-15.  Back to cited text no. 13
    
14.
Parize P, Chandesris MO, Lanternier F, Poirée S, Viard JP, Bienvenu B, et al. Antifungal therapy of Aspergillus invasive otitis externa: Efficacy of voriconazole and review. Antimicrob Agents Chemother 2009;53:1048-53.  Back to cited text no. 14
    
15.
Bagshaw E, Enoch DA, Blackney M, Posthumus J, Kuessner D. Economic impact of treating invasive mold disease with isavuconazole compared with liposomal amphotericin B in the UK. Future Microbiol 2018;13:1283-93.  Back to cited text no. 15
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
 
 
    Tables

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