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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 21  |  Issue : 4  |  Page : 282-286

Histologically confirmed ocular tuberculosis: an unusual case and review of literature


1 Department of Infectious Diseases, Hedi Chaker University Hospital, University of Sfax, Tunisia
2 Department of Ophthalmology, Habib Bourguiba University Hospital, University of Sfax, Sfax, Tunisia
3 Department of Pathology, Habib Bourguiba University Hospital, University of Sfax, Sfax, Tunisia

Date of Submission18-Mar-2020
Date of Decision24-Apr-2020
Date of Acceptance20-Jun-2020
Date of Web Publication28-Dec-2020

Correspondence Address:
MD Makram Koubaa
Department of Infectious Diseases, Hedi Chaker University Hospital, University of Sfax, Sfax, 3029
Tunisia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/DJO.DJO_21_20

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  Abstract 


Ocular tuberculosis (OTB) is the first cause of ocular inflammation of infectious origin in developing countries. It can affect all structures of the eye without pathognomonic presentation. Its diagnosis remains challenging owing to the difficulty of sampling for histopathological and microbiological investigations. A 36-year-old male patient with no previous medical history was admitted for a scleral mass in his right eye, which was diagnosed elsewhere 45 days before. On examination, a minimal intraocular inflammatory reaction and right eyelid swelling were noted. A scleral lesion in the superior nasal quadrant of the right eye was observed and was responsible for globe displacement inferolaterally and for restricted eye motility. An excision biopsy of the suspicious lesion was made. Histological examination revealed a tuberculous granuloma associated with caseous necrosis. Sputum smear result was negative for Mycobacterium tuberculosis. Further investigations excluded extra-OTB involvement. The patient received 6 months of antitubercular therapy. The disease evolution was favorable. The authors also reviewed all reported cases of histologically confirmed OTB in the literature by search in PubMed till December 2019. Cases with histopathology results revealing granulomatous inflammation without caseous necrosis and cases of tubercular dacryoadenitis were excluded from the search, at enrollment.

Keywords: antitubercular therapy, caseous necrosis, ocular tuberculosis, scleral mass


How to cite this article:
Hammami F, Koubaa M, Bougharriou I, Gargouri S, Charfi S, Boudawara T, Feki J, Jemaa MB. Histologically confirmed ocular tuberculosis: an unusual case and review of literature. Delta J Ophthalmol 2020;21:282-6

How to cite this URL:
Hammami F, Koubaa M, Bougharriou I, Gargouri S, Charfi S, Boudawara T, Feki J, Jemaa MB. Histologically confirmed ocular tuberculosis: an unusual case and review of literature. Delta J Ophthalmol [serial online] 2020 [cited 2022 Jun 27];21:282-6. Available from: http://www.djo.eg.net/text.asp?2020/21/4/282/305284




  Introduction Top


Tuberculosis has been declared as a global emergency by the WHO, as it remains the most common cause of mortality from any single infectious disease [1]. It is a multisystem disease with a wide spectrum of clinical presentations. Ocular tuberculosis (OTB) is involved in 5–10% of ocular inflammation cases. It can affect all structures of the eye without pathognomonic presentation [2]. The most common presentations of OTB are anterior uveitis, choroiditis, and sclerokeratitis [3]. OTB is the first cause of ocular inflammation of infectious origin in developing countries, representing 60% of infectious uveitis and 5% of ocular inflammation of all etiologies combined [4]. The disease remains challenging owing to the lack of clear guidance on diagnosis, investigation, and management [5]. In fact, the diagnosis confirmation by histopathologic staining methods of affected tissues or by standard microbiological methods such as demonstration of Mycobacterium tuberculosis organisms on smear microscopy or culture from ocular samples is usually difficult owing to the paucibacillary nature of disease in the eye [6].

Here, we report a case of OTB in a previously healthy 36-year-old male patient, and we reviewed all reported cases of histologically confirmed OTB in the literature by a search in PubMed till December 2019. Cases of OTB with histopathology results revealing granulomatous inflammation without caseous necrosis and cases of tubercular dacryoadenitis were excluded from the search, at enrollment. Written consent to publish the case report has been obtained from the patient.


  Case presentation Top


A 36-year-old male patient with no previous medical history was admitted for a scleral mass in his right eye, which was diagnosed elsewhere 45 days before. The patient had no complaints such as pain, redness, or photophobia. On examination, a minimal intraocular inflammatory reaction and right eyelid swelling were noted. A scleral lesion in the superior nasal quadrant of the right eye was observed and was responsible for globe displacement inferolaterally and for restricted eye motility. Intraocular pressure was 12 and 14 mmHg in the right and left eye, respectively. The visual acuity was 9/10 and fundus was normal in both eyes. Left eye anterior and posterior segment examination was within normal limits.

Laboratory investigations revealed hemoglobin level of 13.4 g/dl, leukocyte count of 5500 cells/mm3, and platelets count of 162 000/mm3. Erythrocyte sedimentation rate was 40 mm, and C-reactive protein was 12 mg/l. Hepatic and renal functions were both normal. Fluorescein angiography confirmed our examination results by showing a lesion of the right globe. HIV and hepatitis B and C serologic test results were negative. There was no remarkable finding on chest radiography. Cerebral and orbital MRI showed an enhancement of the scleral lesion associated with a major inflammatory reaction surrounding the right globe ([Figure 1]).
Figure 1 Frontal T1-weighted post-gadolinium magnetic resonance imaging showing homogeneous enhancement of the scleral lesion, measuring 12 mm, localized in the superior nasal quadrant of the right eye, associated with an extraocular enhancement (arrow).

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An excision biopsy of the suspicious lesion was made. The specimen was subjected to microscopic examination and culture for bacteria, mycobacteria, and fungi. Cultures remained sterile after 2 weeks. Histological examination revealed a tuberculous granuloma associated with caseous necrosis and absence of signs of malignancy ([Figure 2]). The diagnosis of OTB was therefore confirmed. Sputum smear was negative for Mycobacterium tuberculosis. QuantiFERON TB gold test result was negative. A thoracoabdominal computed tomography scan result was normal and showed no evidence of tuberculosis elsewhere.
Figure 2 Histopathology of the mass revealing granulomatous inflammation (arrows) associated with a focus of caseous necrosis (star) (hematoxylin and eosin, ×100).

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The patient was treated with antitubercular therapy starting with fixed-dose combination drugs including isoniazid, rifampin, pyrazinamide, and ethambutol for the first 2 months, followed by isoniazid-rifampicin bitherapy for the next 4 months. The treatment was well tolerated. The disease evolution was favorable, and the patient remained asymptomatic after a 2-year follow-up. The visual acuity remained 9/10 after completion of antitubercular therapy.


  Discussion Top


The literature search yielded 19 cases of histopathologically confirmed OTB, showing both granulomatous inflammation and caseous necrosis, including our case [3] ([Table 1]). Biopsy or excision of an orbital/eyelid mass or nodule was enough to confirm the diagnosis in most cases. However, enucleation was indicated in three cases [6],[7],[10]. Extraocular involvement was rarely noted [7],[10],[17],[18],[20], among which pulmonary tuberculosis was the most common associated site [7],[10],[17],[20].
Table 1 Summary of all reported patients with histologically confirmed ocular tuberculosis

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The diagnosis of OTB remains challenging, as the commonly used laboratory tests that also serve as the gold standard for confirming the diagnosis of tuberculosis, namely, the demonstration of Mycobacterium tuberculosis organisms by smear or culture in the sputum or the body fluids/tissues, are seldom, if ever, positive from the ocular fluids [21]. Definitive intraocular evidence by PCR needs an invasive diagnostic vitrectomy [22]. Besides, it is further limited by low sensitivity and false positivity [22]. In addition, obtaining proper tissue specimen for histopathological examination is not feasible in most cases [23]. That is why, OTB diagnosis is largely based on a presumptive evidence of systemic infection such as a positive tuberculin skin test result, radiographs showing pulmonary involvement, or positive biopsy of the lymph node [23]. However, the challenge is raised with the lack of evidence of tuberculosis elsewhere in the body. In fact, previous studies reported the absence of evidence for pulmonary tuberculosis in up to 60% of the patients with extrapulmonary tuberculosis, leading to a difficult and delayed diagnosis of the infection [24]. A negative tuberculin skin test result does not rule out the diagnosis of OTB. False-negative reactions are estimated to occur in ∼5–10% of patients. It can be observed early in the infection before hypersensitivity develops, in anergic individuals, in those with severe illness, in new born infants, and in infants or immunosuppressed patients [25].

Different segments of the eye might be involved. These include choroidal lesions, retinal lesions, optic nerve lesions, and intermediate and anterior uveitis [26]. In tuberculosis-endemic areas, broad-based posterior synechiae, retinal vasculitis, and serpiginous-like choroiditis are highly suggestive of OTB [2]. Fluorescein angiography is of great benefit for the evaluation and management of presumed tuberculous retinal vasculitis [25].

Most ocular and systemic investigations are unable to identify Mycobacterium tuberculosis, especially in nonendemic countries. This has led many authors to recommend recovery on the antitubercular therapy regimen as a part of the clinical criteria for diagnosing presumed tuberculosis-related ocular inflammation [2]. To prevent an initial paradoxical worsening, systemic corticosteroids might be used as they help to prevent further damage to ocular tissues from the inflammatory response. However, corticosteroid treatment alone can cause further progression of the infection and so should not be used without concomitant antitubercular therapy [1].


  Conclusion Top


OTB, a rare disease with a myriad presentation, remains a diagnostic challenge. Clinicians must be aware of its clinical manifestations to guide the diagnostic process, shorten the diagnosis delay, and decrease complicated forms.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

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Pirraglia MP, Tortorella P, Abbouda A, Toccaceli F, La Cava M. Spectral domain optical coherence tomography imaging of tubercular chorioretinitis and intraretinal granuloma. Intraretinal tuberculosis: a case report. Int Ophthalmol 2015; 35:445–450.  Back to cited text no. 24
    
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[PUBMED]  [Full text]  


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