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 Table of Contents  
CASE REPORT
Year : 2019  |  Volume : 33  |  Issue : 2  |  Page : 108-111

Isolated tubercular granuloma at orbital apex


1 Department of Otorhinolaryngology, IMS and SUM Hospital, Siksha “O” Anusandhan (Deemed to be University), Bhubaneswar, Odisha, India
2 Department of Ophthalmology, IMS and SUM Hospital, Siksha “O” Anusandhan (Deemed to be University), Bhubaneswar, Odisha, India
3 Medical Research Laboratory, IMS and SUM Hospital, Siksha “O” Anusandhan (Deemed to be University), Bhubaneswar, Odisha, India

Date of Submission13-Feb-2019
Date of Acceptance04-Aug-2019
Date of Web Publication11-Feb-2020

Correspondence Address:
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/jms.jms_10_19

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  Abstract 


Isolated tubercular granuloma at the orbital apex is an extremely rare clinical entity which may lead to difficulties in the diagnosis because of the similarities in the presentation to that of a neoplasm. We are presenting a case of 19-year-old male with isolated tubercular lesion at orbital apex. The diagnosis was confirmed by endoscopic biopsy and treated with antitubercular therapy. The diagnosis of isolated tubercular lesion at the orbital apex mainly depends on the clinician having a high index of suspicion. Although it is extremely rare, tuberculosis can be kept in mind and considered as a differential diagnosis of patients presenting with a solitary tumor at orbital apex to avoid unnecessary surgery.

Keywords: Antitubercular therapy, isolated tubercular granuloma, orbital apex, tuberculosis


How to cite this article:
Swain SK, Das S, Sahu MC. Isolated tubercular granuloma at orbital apex. J Med Soc 2019;33:108-11

How to cite this URL:
Swain SK, Das S, Sahu MC. Isolated tubercular granuloma at orbital apex. J Med Soc [serial online] 2019 [cited 2020 Oct 31];33:108-11. Available from: https://www.jmedsoc.org/text.asp?2019/33/2/108/278076




  Introduction Top


Tuberculosis (TB) is an infectious clinical entity caused by obligate aerobic, acid-fast bacilli Mycobacterium tuberculosis.[1] TB is still prevalent in developing countries. It has varied clinical presentations and may lead to difficulty in diagnosis. TB is a granulomatous disease with wide distribution and varied clinical presentations. The transmission of the TB usually occurs by airborne and lungs are the most common organ to be affected.[2] Extrapulmonary forms of TB accounts for approximately 20% of all active TB and seen in lymph nodes, bones, kidney, and meninges.[3] TB of the lymph node is the most common extrapulmonary form of TB and affects cervical lymph nodes and lymph nodes around the salivary glands. However, tubercular lesion at the orbital apex area is extremely rare. Clinically, it presents with bulging of the eyeball and sometimes pain in the eye with loss of vision. The diagnosis of orbital apex TB needs a high degree of clinical suspicion. If there is no history of pulmonary TB and no relevant symptoms, diagnosis can be extremely difficult. Many times, clinicians do not consider TB in their differential diagnosis of patients presents with otorhinolaryngological symptoms, so often miss the diagnosis and lead to inappropriate management. A high index of suspicion is required when a patient presents with otorhinolaryngological TB as it often leads an indolent course and its presentations vary according to the different anatomical sites and adjacent areas. Here, we are presenting a case of the isolated tubercular lesion at orbital apex of a 19-year-old patient which is an extremely rare clinical entity in medical literature.


  Case Report Top


A 19-year-old boy attended at the outpatient department of otorhinolaryngology with complaints of protrusion of the right eye [Figure 1] with mild pain since 3 months. The clinical presentations are slowly progressive in nature. There was no comorbidities and associated with no previous history of trauma or surgery. The ophthalmological examination showed normal conjunctiva, cornea, iris, pupil, anterior chamber, and lens. The orbital margins were free and no obvious, palpable mass. Fundoscopy revealed blurring at the disc margin. There were normal visual acuity and extraocular movements. Examinations of nose and paranasal sinuses were within normal limit. There was no obvious mass seen during anterior rhinoscopy. Diagnostic nasal endoscopy showed a mild bulging posteriorly medial to the middle turbinate. Magnetic resonance imaging (MRI) scan of the orbit (axial view) was done which showed a soft-tissue mass at the orbital apex [Figure 2]. The computed tomography scan revealed a small lesion at the orbital apex with proptosis of the right eyeball [Figure 3]. Blood investigations showed raised erythrocyte sedimentation rate (ESR) of 65/h and other routine blood tests were within normal limit. His chest X-ray and lumbar puncture were normal. Investigations revealed a strongly positive Mantoux test (>15 mm erythema within 24 h). The patient was planned for endoscopic biopsy under general anesthesia. Right uncinectomy, anterior and posterior ethmoidectomy done then sphenoidotomy were done. Following these surgical steps, the area adjacent to optic nerve was exposed and mass at the orbital apex was carefully debulked [Figure 4] and sent for the histopathological report. It revealed caseating granuloma in the histopathological report [Figure 5] and positive QuantiFERON-TB Gold test. He was commenced on antituberculous therapy. Anti-TB therapy was given for 6-month duration with 2 months of intensive phase. A standard treatment regime of ethambutol, isoniazid, rifampicin, and pyrazinamide was prescribed for 2 months followed by 4-month course of rifampicin and isoniazid. After the treatment course of 2 months, the right eye reduced significantly. Review after 6 months and 1 year of treatment, did not reveal any recurrence of symptoms.
Figure 1: Patient presenting with right eye proptosis

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Figure 2: Magnetic resonance imaging of the orbit showing a soft-tissue mass at the right orbital apex

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Figure 3: Plain computed tomography of the paranasal sinus showing a retro-orbital mass in the right orbit

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Figure 4: Intra-operative endoscopic picture adjacent to the right side orbital apex

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Figure 5: (a) Photomicrograph of histopathological report showing aggregations of epithelioid cells and peripheral lymphocytes forming a circumscribed with caseous necrosis (H and E, ×40) (b) Ziehl–Nielsen staining from biopsy specimen showing acid-fast bacilli

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  Discussion Top


TB is a chronic granulomatous infectious disease of the human being known since Hippocrates period. It is an airborne infectious disease caused by obligate aerobic, acid-fast bacilli M. tuberculosis.[4] It most commonly affects the lungs. TB has a worldwide presence, and no part of the human body is immune to it. It is a common disease seen in developing country like India. Approximately 10% of the TB case affects the head and neck region.[5] Its incidence is increasing with the prevalence of HIV infection. TB of the orbit is a rare clinical entity whereas TB at orbital apex is an extremely rare and often a potentially devastating condition if not treated early can lead into grave situation.[6] Protrusion of the eyeball secondary to endocrine causes is called exophthalmos whereas nonendocrine as proptosis. In adult population, thyroid orbitopathy is the most common etiology for bilateral as well as unilateral exophthalmos. Other etiologies are nontubercular infections, inflammatory conditions such as pseudotumor and neoplasms as cavernous hemangiomas, lymphangiomas, lymphoma, and Wegener's granulomatosis. The diagnosis of the tubercular lesion at the orbital apex is often difficult as it can affect any part of the orbit and the clinical presentations often mimic a wide range of different underlying diseases which require complete workup. Delayed diagnosis and treatment lead to significant vision loss.[7] The exact diagnosis needs a high index of suspicion, assessment of clinical presentations, and appropriate laboratory findings. There are no standard criteria for the diagnosis of the TB at orbital apex, but there are several factors can help to diagnose the disease.[8] The diagnosis is confirmed by tissue diagnosis. Proptosis due to space occupying lesion at the orbital apex can lead to compromised visual function, papillary dysfunction, color blindness, and constriction of the visual field due to optic nerve compression. Proptosis may also lead to exposure keratopathy. TB should be anticipated in patients with retro-orbital soft-tissue mass if the patient comes from endemic areas or having systemic or constitutional symptoms. These will help for early diagnosis and treatment. A delay in diagnosis may lead to optic nerve compression and likely to poor recovery. As the clinical presentations often mimic to signs and symptoms of an inflammatory optic neuropathy, so this case may be confused with demyelinating optic neuropathy. However, patients coming from TB endemic region and presenting constitutional symptoms, proptosis, and affecting other cranial nerves those are passing through the orbital apex are strong indicators of alternate diagnosis. TB at the orbital apex may lead to orbital apex syndrome which is defined as an optic neuropathy and/or ophthalmoplegia and loss of sensation due to the lesion at the orbital apex by affecting multiple cranial nerves.[9] The tubercular granulomatous lesion at the orbital apex has good prognosis if treated early. The progression of this lesion due to delayed or without treatment leads to loss of corneal sensation, sluggish or absent papillary response to light, blurring of the vision, exophthalmos, complete ophthalmoplegia, and blindness due to occlusion of retinal and ciliary artery. There is a chance of intracranial extension and formation of tuberculoma in the brain. The use of tuberculin skin or Mantoux test can provide variable information, but it provides an initial suspicion. The definitive diagnosis of TB depends on the isolation of M. tuberculosis from the diagnostic specimen. Other investigations include blood tests such as complete blood count, ESR, liver function test, chest X-ray, orbital and brain imaging (MRI), and lumbar puncture. In our case, the patient was taken up for endoscopic biopsy from the orbital apex under general anesthesia. Right uncinectomy, anterior and posterior ethmoidectomy were done then sphenoidotomy was done. Following these surgical steps, optic nerve decompression was done, and mass at the orbital apex was carefully debulked and sent for histopathological examination. The mainstay of treatment is medical, and it includes antitubercular therapy for at least 6 months. The treatment of TB at the orbital apex includes 2 months' course of rifampicin, isoniazid, pyrazinamide, and ethambutol followed by a 4 months' course of isoniazid and rifampicin.[10] The effective antitubercular therapy can lead to resolution of the lesion and avoid unnecessary surgery. In case of TB of orbital apex and impaired visual acuity, the combination of anti-TB treatment and high dose of corticosteroids are highly useful for restoring visual acuity. The use of corticosteroids is helpful to treat inflammatory component of the disease and prevent a Jarisch–Herxheimer type reaction on common antibiotics. Orbital apex TB is difficult to diagnose as it can mimic too many more pathologies including neoplasm. A high index of suspicion for TB at orbital apex and need of biopsy with the cooperation of infectious disease specialists are paramount, as timely diagnosis and treatment may prevent irreversible vision loss and spread of disease.[11]


  Conclusion Top


Successful treatment of tubercular lesions at the orbital apex area requires prompt diagnosis, and rapid institution of antitubercular therapy as delay or nonaggressive treatment can lead into spread of the infection with lethal consequences. Radiological imaging is an important diagnostic test in this lesion for early diagnosis and directing the treatment. Endoscopic clearance of the disease and combined with antitubercular therapy appears to be an appropriate modality of the treatment.

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.
Tabbara KF. Tuberculosis. Curr Opin Ophthalmol 2007;18:493-501.  Back to cited text no. 1
    
2.
Jones RM, Brosseau LM. Aerosol transmission of infectious disease. J Occup Environ Med 2015;57:501-8.  Back to cited text no. 2
    
3.
Lee IK, Liu JW. Tuberculous parotitis: Case report and literature review. Ann Otol Rhinol Laryngol 2005;114:547-51.  Back to cited text no. 3
    
4.
Swain SK, Behera IC, Sahu MC. Primary sinonasal tuberculosis: Our experiences in a tertiary care hospital of Eastern India. Egypt J Ear Nose Throat Allied Sci 2017;18:237-40.  Back to cited text no. 4
    
5.
Menon K, Bem C, Gouldesbrough D, Strachan DR. A clinical review of 128 cases of head and neck tuberculosis presenting over a 10-year period in Bradford, UK. J Laryngol Otol 2007;121:362-8.  Back to cited text no. 5
    
6.
Ha JK, Park BJ, Kim YH, Lim YJ. Orbital solitary fibrous tumor: A case report and diagnostic clues. J Korean Neurosurg Soc 2009;46:77-80.  Back to cited text no. 6
    
7.
Patel SS, Saraiya NV, Tessler HH, Goldstein DA. Mycobacterial ocular inflammation: Delay in diagnosis and other factors impacting morbidity. JAMA Ophthalmol 2013;131:752-8.  Back to cited text no. 7
    
8.
Sharma A, Thapa B, Lavaju P. Ocular tuberculosis: An update. Nepal J Ophthalmol 2011;3:52-67.  Back to cited text no. 8
    
9.
Hughes EH, Petrushkin H, Sibtain NA, Stanford MR, Plant GT, Graham EM. Tuberculous orbital apex syndromes. Br J Ophthalmol 2008;92:1511-7.  Back to cited text no. 9
    
10.
Center for Disease Control. Core Curriculum on Tuberculosis: What Clinicians Should Know. 6th ed. Center for Disease Control; 2013. Available from: http://www.cdc.gov/tb/education/corecurr/pdf/chapter 6.pdf. [Last accessed on 2016 Mar 20].  Back to cited text no. 10
    
11.
Thompson MJ, Albert DM. Ocular tuberculosis. Arch Ophthalmol 2005;123:844-9.  Back to cited text no. 11
    


    Figures

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



 

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