Latent Mycobacterial fortuitum infection presenting as ocular sarcoidosis

Kalpana Babu; Preethi Hegde; Suma P Kumar

doi:10.4103/ijo.IJO_1370_22

CASE REPORT

Year : 2023 | Volume : 3 | Issue : 2 | Page : 513-515

Latent Mycobacterial fortuitum infection presenting as ocular sarcoidosis

Kalpana Babu¹, Preethi Hegde¹, Suma P Kumar²
¹ Department of Uvea and Ocular Inflammation, Prabha Eye Clinic and Research Centre and Vittala International Institute of Ophthalmology, Bengaluru, Karnataka, India
² Department of Respiratory Medicine, Excel Care Hospitals, Bengaluru, Karnataka, India

Date of Submission	08-Jun-2022
Date of Acceptance	07-Dec-2022
Date of Web Publication	28-Apr-2023

Correspondence Address:
Kalpana Babu
504, 40^th Cross, Jayanagar 8^th Block, Bengaluru - 560 070, Karnataka
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_1370_22

Abstract

A 62-year-old lady presented with bilateral granulomatous uveitis and cystoid macular edema. A diagnosis of ocular sarcoidosis was made (revised International Workshop on Ocular Sarcoidosis [IWOS] criteria) on the basis of lymphopenia, negative Mantoux, and QuantiFERON TB gold tests. Enlarged mediastinal lymph nodes, subpleural nodules, and nodular interstitial thickening were seen on high-resolution computed tomography (HRCT) thorax. Non-necrotizing granulomato us inflammation, negative for mycobacteria on staining and GeneXpert, was noted on endobronchial ultrasound-guided transbronchial lymph node aspiration (EBUS-TBNA). However, culture grew Mycobacterium fortuitum after 3 weeks. This case highlights a rare presentation of latent M. fortuitum infection presenting as ocular sarcoidosis, speculating the coexistence of two diseases in an individual, one possibly triggering the other.

Keywords: Atypical mycobacteria, EBUS-TBNA, Mycobacterium fortuitum, ocular sarcoid, uveitis

How to cite this article:
Babu K, Hegde P, Kumar SP. Latent Mycobacterial fortuitum infection presenting as ocular sarcoidosis. Indian J Ophthalmol Case Rep 2023;3:513-5

How to cite this URL:
Babu K, Hegde P, Kumar SP. Latent Mycobacterial fortuitum infection presenting as ocular sarcoidosis. Indian J Ophthalmol Case Rep [serial online] 2023 [cited 2023 Jun 2];3:513-5. Available from: https://www.ijoreports.in/text.asp?2023/3/2/513/374894

We report a rare presentation of latent Mycobacterial fortuitum infection presenting as ocular sarcoidosis.

Case Report

A 62-year-old lady was referred to us for diminution of vision in both eyes of 5 months duration. She had a history of an uneventful phacoemulsification elsewhere with intraocular lens implantation in the left eye 5 months ago. She was on topical interferon drops, started by her ophthalmologist, for a probable diagnosis of Irvine Gas syndrome. Recently, she had been using topical prednisolone acetate eye drops four times/day for redness and pain in the right eye for 1 week. Systemic history was significant for diabetes mellitus and hypertension. She was on telmisartan for hypertension.

On examination, her best-corrected visual acuity (BCVA) was 6/60 and 6/18 in the right and left eyes, respectively. Right eye examination showed a few medium-sized keratic precipitates, broad-based posterior synechiae of 1–2 clock hours, an anterior chamber reaction 1+ (Standardization of Uveitis Nomenclature [SUN]),^[1] a posterior subcapsular cataract, few vitreous cells, and cystoid macular edema. Left eye examination showed a quiet anterior segment, few vitreous cells, and cystoid macular edema [Figure 1]. Fluorescein angiography showed diffuse retinal vascular leakages with a petaloid pattern in the fovea in both eyes. Indocyanine green angiography did not show any choroidal granulomas. Optic coherence tomography (OCT) showed cystoid macular edema [Figure 2]. Laboratory investigations showed decreased lymphocyte counts on complete hemogram (17%, range: 20%–40%). Erythrocyte sedimentation rate (ESR) was 28 mm/h. Mantoux, QuantiFERON TB gold, Venereal Disease Research Laboratory (VDRL), and Treponema pallidum hemagglutination (TPHA) tests were negative. Serum angiotensin-converting enzyme was in the normal range (57 U/L, normal range: 12–68 U/L). Random blood sugar, urine routine, serum calcium, liver function, and renal function tests were normal. High-resolution computed tomography (HRCT) thorax showed multiple enlarged mediastinal lymph nodes, nodular interstitial thickening in bilateral lower lobes, right middle lobe, and lingular segment of the left upper lobe, and few centrilobular and subpleural nodules in the right lung [Figure 3]. Endobronchial ultrasound-guided transbronchial lymph node aspiration (EBUS-TBNA) showed non-necrotizing granulomatous inflammation. Staining for acid-fast bacilli (AFB) and GeneXpert/rifampicin resistance for mycobacteria were negative. Based on the revised International Workshop on Ocular Sarcoidosis (IWOS) criteria,^[2] she was diagnosed to have ocular sarcoidosis and started on oral steroids. However, 3 weeks later, the culture grew Mycobacteria fortuitum. She was started on intravenous amikacin 1000 mg for 5 days, tablet linezolid 600 mg twice a day, and tablet moxifloxacin 400 mg once a day by the pulmonologist. At 5 months follow-up, she is doing well with no recurrences of inflammation. Her BCVA was 6/9 (OU) [Figure 4].

Figure 1: Composite slit-lamp photographs of the right eye (a) showing medium-sized keratic precipitates (yellow arrow) and broad posterior synechiae and of the left eye (b) showing pseudophakia and quiet anterior segment. Fundus photographs showing hazy view with only disk seen in the right eye (c) and cystoid macular edema in the left eye (d)

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Figure 2: Composite fluorescein and ICG angiography photographs showing diffuse retinal vascular leakage with petaloid appearance in right (a) and left (b) eyes. Spectral OCT showing cystoid macular edema in right (c) and left (d) eyes. ICG = indocyanine green, OCT = optical coherence tomography

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Figure 3: High-resolution computed tomography shows mediastinal lymph node enlargements (a and b, yellow arrows) and subpleural and septal thickening (c and d, yellow circles)

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Figure 4: Serial OCT photographs of the right (a) and left (b) eyes at 1, 2, and 3 months showing the resolution of cystoid macular edema with treatment. OCT = optical coherence tomography

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Discussion

Sarcoidosis is a chronic, multisystem, granulomatous disease which occurs as a result of an exaggerated cellular immune response to self- or non–self-antigens in a genetically susceptible individual.^[3] Although the etiology is unclear, environmental stimuli (asbestos, tattoo induced), infections such as mycobacteria and Propionibacterium acnes, drugs like checkpoint inhibitors, and malignancies such as lymphoma are associated with development of sarcoidosis.^[4],[5] The diagnosis is made on the basis of a triad of typical clinical features, positive chest radiology findings, and non-necrotizing granulomatous inflammation on biopsy. This disease is similar to tuberculosis clinically, radiologically, and histopathologically. In addition, recent evidence suggesting a strong mycobacterial link in its pathogenesis poses a significant diagnostic challenge in high TB-endemic countries like India.^[6] Ocular involvement may be the initial or the only manifestation of sarcoidosis. The revised IWOS^[2] criteria are useful in identifying cases of ocular sarcoidosis. Based on these criteria, that is, lymphopenia, negative Mantoux and QuantiFERON TB gold tests, and consistent chest radiology findings on HRCT and EBUS-TBNA showing non-necrotizing granulomas negative for mycobacteria on staining, GeneXpert, and polymerase chain reaction (PCR), our patient was diagnosed to have definite ocular sarcoidosis. Serum angiotensin converting enzyme (ACE) was in the normal range, probably due to the ACE inhibitors she was taking for her hypertension. But 3 weeks later, the culture of the TBNA aspirate grew Mycobacterium fortuitum. This case is interesting because of the existence of culture-proven latent atypical mycobacterial infection in a patient with ocular sarcoid. We have often seen Mycobacterium tuberculosis (MTb) complex associated with sarcoid in a high TB-endemic country like India. This is a rare occurrence of atypical mycobacterial infection (M. fortuitum) in a patient with ocular sarcoidosis. A meta-analysis by Gupta et al.^[6] concluded that an association between mycobacteria (both MTb complex and nontuberculous) and sarcoidosis exists, and even suggested that these mycobacteria might be members of slow-growing types with low pathogenic potential, but had a capability of eliciting a type IV immune response. We speculate that the atypical M. fortuitum infection could have triggered the development of sarcoidosis in our case.

Conclusion

A rare presentation of latent M. fortuitum infection presenting as ocular sarcoid is highlighted in this case report, speculating the coexistence of two diseases in an individual, one possibly triggering the other.

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

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