|Year : 2023 | Volume
| 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|
504, 40th Cross, Jayanagar 8th Block, Bengaluru - 560 070, Karnataka
Source of Support: None, Conflict of Interest: None
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]), 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, 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)|
Click here to view
|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|
Click here to view
|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)|
Click here to view
|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|
Click here to view
| 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. 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., 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. Ocular involvement may be the initial or the only manifestation of sarcoidosis. The revised IWOS 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. 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
Conflicts of interest
There are no conflicts of interest.
| References|| |
Jabs DA, Nussenblatt RB, Rosenbaum JT; Standardization of Uveitis Nomenclature (SUN) Working Group. Standardization of uveitis nomenclature for reporting clinical data. Results of the first international workshop. Am J Ophthalmol 2005;140:509-16.
Mochizuki M, Smith JR, Takase H, Kaburaki T, Acharya NR, Rao NA; International Workshop on Ocular Sarcoidosis Study Group. Revised criteria of international workshop on ocular sarcoidosis (IWOS) for the diagnosis of ocular sarcoidosis. Br J Ophthalmol 2019;103:1418-22.
Rothova A. Ocular involvement in sarcoidosis. Br J Ophthalmol 2000;84:110-6.
Babu K. Sarcoidosis in tuberculosis endemic regions: India. J Ophthalmic Inflamm Infect 2013;3:53.
Judson MA. Environmental risk factors for sarcoidosis. Front Immunol 2020;11:1340.
Gupta D, Agarwal R, Aggarwal AN, Jindal SK. Molecular evidence for the role of mycobacteria in sarcoidosis: A meta-analysis. Eur Respir J 2007;30:508-16.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]