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Year : 2020  |  Volume : 9  |  Issue : 3  |  Page : 319-321

Tuberculous otitis media with osteomyelitis of the regional craniofacial bones

Department of Otorhinolaryngology, Cho Ray Hospital, Ho Chi Minh City, Vietnam

Date of Submission23-Jun-2020
Date of Decision01-Jul-2020
Date of Acceptance01-Jul-2020
Date of Web Publication28-Aug-2020

Correspondence Address:
Tran Minh Truong
Department of Otorhinolaryngology, Cho Ray Hospital, Ho Chi Minh City
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijmy.ijmy_112_20

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Tuberculosis is an uncommon cause of chronic suppurative otitis media. Delay in diagnosis can lead to delayed treatment, resulting in significant complications. We describe a case of tuberculous otitis media with osteomyelitis of the craniofacial bones in a 44-year-old woman with chronic painless suppurative otorrhea, not responding to antibiotics, hearing loss, and facial palsy. Chest X-ray was normal. Computed tomography of the temporal bone showed the destruction of the left zygomatic bone, clivus, and petrous part of the temporal bone. Polymerase chain reaction was positive for Mycobacterium tuberculosis (MTB), and histopathological findings showed caseous necrotizing tissues. Sputum culture was negative for MTB. The patient was successfully treated with surgery and anti-tuberculosis drugs. The polymerase chain reaction is a sensitive, rapid diagnostic tool used to diagnose TB. Surgical approaches and operative biopsy should be considered when the cause of the chronic purulent discharge is still unknown.

Keywords: Chronic otorrhea, craniofacial osteomyelitis, tuberculous otitis media

How to cite this article:
Truong TM, Uyen TH. Tuberculous otitis media with osteomyelitis of the regional craniofacial bones. Int J Mycobacteriol 2020;9:319-21

How to cite this URL:
Truong TM, Uyen TH. Tuberculous otitis media with osteomyelitis of the regional craniofacial bones. Int J Mycobacteriol [serial online] 2020 [cited 2021 Jan 20];9:319-21. Available from: https://www.ijmyco.org/text.asp?2020/9/3/319/293537

  Introduction Top

Tuberculosis commonly affects the lungs but can involve any organ of the body. Tuberculosis involving the ear and mastoid is the rarest form of extrapulmonary tuberculosis.[1] It accounts for 0.05%–0.9% of all cases of chronic otitis media (COM).[2] To the best of our knowledge, this is the first case of aural tuberculosis with osteomyelitis of the regional craniofacial bones reported in the literature. Herein, we describe the clinical features of tuberculous otitis media (TOM), emphasize the importance of the polymerase chain reaction (PCR), and operative biopsy in the diagnosis of this disease.

  Case Report Top

A 44-year-old female presented to our hospital because of chronic otorrhea, ipsilateral facial palsy, and hearing loss. She had a history of COM with recurrent meningitis and had been treated with ceftazidime for >3 weeks, but the symptoms did not improve.

On physical examination, she had evidence of nuchal rigidity. Kernig's sign was positive. Lungs were clear, sounding to auscultation. The purulent fluid drained from the left ear canal. There were also fistulous tracts in the mastoid and zygomatic area. Pale granulation was seen in the external ear canal [Figure 1].
Figure 1: Purulent discharge with pale granulation in the external ear canal

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Endoscopy showed a large tympanic membrane perforation, pus with granulation in the external ear canal and middle ear cavity [Figure 2], and purulent discharge from the left sphenoid sinus. Imaging with computed tomography (CT) showed erosion of the left zygomatic bone, maxillary bone, clivus, and petrous part of the temporal bone [Figure 3]. The chest radiograph appeared normal. A sputum smear was negative for tuberculosis on staining and proved to be culture-negative weeks later.
Figure 2: After antituberculous therapy, the wound had not healed completely, but the ear discharge stopped

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Figure 3: Destruction of left zygomatic, temporal, maxillary, clivus, and sphenoid bones

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All of the samples, including bones and tissues from the sphenoid sinus, mastoid, zygomatic, and maxillary bones, were sent for culture and PCR for Mycobacterium tuberculosis (MTB). All of which were positive for tuberculosis. Cultures from the zygomatic and maxillary bone showed positive results for Mycobacterial species, whereas the other samples showed negative results. Microscopy of the biopsy material showed caseous necrotizing granulomas.

Treatment was started with antituberculous drugs, and localized surgery was undertaken. During surgery, the affected area showed pale granulation tissue and copious pus filling the mastoid cavity and the external ear canal. There was the destruction of the mastoid bone, with an extension of disease from the external ear canal medially to the apex of the temporal bone, exposing the internal carotid artery. The zygomatic and maxillary bone was soft, showing necrotic tissue with debridement. All necrotic bone was removed.

The patient was subsequently started on anti-tuberculosis therapy, including rifampicin, isoniazid, pyrazinamide, and ethambutol.

Symptoms dramatically improved within 2 weeks. The patient continued with triple anti-tuberculosis therapy for 9 months. Follow-up after 1 year showed no evidence of purulent discharge.

  Discussion Top

In 1908, Bezold first described zygomatic abscess due to otitis media as an exceedingly rare occurrence. The disease usually occurs in the well pneumatized zygomatic root with ample zygomatic root cells, which are contiguous with the rest of the mastoid cell system.[3] Due to this unique structure, pathogens from the temporal bone can spread to the facial bone and skull.

Pathogens can infect the ear by three pathways: via the bloodstream (hematogenous spread), lymphatic vessels, or locally via the Eustachian tube. The involvement of the mastoid and ear is hypothesized to be of hematogenous origin.[4],[5]

The classical symptoms include tympanic membrane perforation, chronic purulent otorrhea that does not respond to systemic and topical antibiotics, facial paralysis, and hearing loss. At an advanced stage, granulation tissue can erode the ossicles and block the aditus ad-antrum, filling the mastoid cavity and extending it to the external ear canal. With further progression, it can result in osteomyelitis of the temporal bone. In our case, the disease caused destruction and osteomyelitis of the regional craniofacial bones.

Utilizing all the information from the patient's signs and symptoms, laboratory tests, radiologic studies (CT scan), and surgical pathology, we successfully differentiated TOM from COM. Key clinical indicators included chronic otorrhea not responsive to routine antibiotic administration, large tympanic membrane perforation with pus in the middle ear, and CT findings of bone erosion in the affected region. The surgical pathological findings, including caseous necrotic bone and pale granulation tissue in the tympanic cavity and mastoid area, were important clues. All PCR samples were positive for MTB, and biopsy showed caseous necrotizing granulomas.

For diagnosis, positive culture and smears for MTB classically need long duration and low yield. Overgrowth of common flora, namely Staphylococcus, Pseudomonas, Klebsiella, Proteus, and Streptococcus could inhibit the growth of MTB. Histopathological findings may show caseous necrotizing, epithelioid granulomas, and few Langhan's giant cells, suggestive of tuberculosis. PCR for MTB is an emerging, rapid diagnostic test with high sensitivity. It can aid in optimizing the treatment of patients with chronic suppurative aural discharge. Indeed, demonstrating MTB in histological samples by classic means obtained during surgery is difficult. Thus, multiple biopsies obtained at multiple sites should increase the yield.

Delayed diagnosis can lead to catastrophic and irreversible damage to the labyrinth and underlying bones of the affected region with hearing loss, scarring, and disfigurement. Current trends in practice suggest antituberculous therapy should be initiated early, based on clinical or suspicious histopathological findings rather than waiting weeks for a culture report.[1] Surgery has a crucial role in abscess drainage and removal of the bone sequestrum. In this case, the patient was diagnosed with TOM by intraoperative biopsy. Findings such as sclerotic changes of the mastoid on imaging, or granulation tissue in the tympanic cavity on the inspection are suggestive features of tuberculosis in the high-risk patient.

  Conclusion Top

TOM is an uncommon but debilitating disease that can lead to hearing loss and localized bony destruction with complications. As tuberculous culture results often take weeks for results, rapid diagnosis such as PCR offer hope. Surgical approaches with intraoperative analyses can be useful. Operative biopsy should be considered when the cause of the chronic purulent discharge is still unknown. Ultimately, the prevention of disease progression is necessary to avoid the most serious complications.

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.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Singh A, Irugu DVK, Verma H, Thakar A. Atypical presentation of aural tuberculosis with complication. BMJ Case Rep 2018;2018:bcr-2017.  Back to cited text no. 1
Awan MS, Salahuddin I. Tuberculous otitis media: Two case reports and literature review. Ear Nose Throat J 2002;81:792-4.  Back to cited text no. 2
Spiegel JH, Lustig LR, Lee KC, Murr AH, Schindler RA. Contemporary presentation and management of a spectrum of mastoid abscesses. Laryngoscope 1998;108:822-8.  Back to cited text no. 3
Adhikari P. Tuberculous otitis media: A review of literature. J The Internet J Otorhinolaryngol 2009;9:7.  Back to cited text no. 4
Adhikari P, Guragain R, Bhusal C. Tuberculous otitis media with facial palsy: A case report. J Nepal J ENT Head Neck Surg 2010;1:17-8.  Back to cited text no. 5


  [Figure 1], [Figure 2], [Figure 3]


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