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      Vanishing retinal arterial aneurysms with anti-tubercular treatment in a patient presenting with idiopathic retinal vasculitis, aneurysms, and neuroretinitis

      case-report

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          Abstract

          Background

          Idiopathic retinal vasculitis, aneurysms, and neuroretinitis (IRVAN) syndrome presents with characteristic clinical manifestations such as aneurysms at arteriolar bifurcations and optic nerve and retinal vascular inflammation. Regression of such features on treatment with anti-tubercular therapy (ATT) combined with corticosteroids has not been reported in literature.

          Findings

          A 30-year-old female with sudden painless decreased vision in the left eye was referred with a diagnosis of presumed tuberculous retinal vasculitis and a positive tuberculin skin test. Based on the clinical and angiographic features of the right eye, a diagnosis of IRVAN syndrome was made. In the left eye, the patient had vitreous hemorrhage for which pars plana vitrectomy was performed. The vitreous sample was positive for Mycobacterium tuberculosis using multiplex polymerase chain reaction, and the patient was started on standard four-drug ATT and oral corticosteroids. At 6-month follow-up, vanishing of retinal arterial aneurysms was observed.

          Conclusions

          The pathogenesis of IRVAN syndrome is uncertain. One of the postulates is that the features of arterial aneurysms and other retinal vascular alterations occur secondary to acquired inflammatory reaction. We hypothesize that IRVAN syndrome may be a morphological diagnosis possibly associated with various entities, one of which could be ocular tuberculosis. It may be prudent to rule out intraocular tuberculosis in cases labeled as IRVAN syndrome in an endemic population.

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          Most cited references15

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          Polymerase chain reaction for detection of Mycobacterium tuberculosis in epiretinal membrane in Eales' disease.

          Tuberculous etiology has been suggested in Eales' disease. Because epiretinal membrane (ERM) is formed on the inner surface of the retina in Eales' disease, it could be the most appropriate intraocular specimen for investigation. Therefore, a nested polymerase chain reaction (nPCR), which detects MPB64 gene of Mycobacterium tuberculosis on the archival specimens of ERM of well-documented Eales' and non-Eales' patients, was applied and the results compared. nPCR technique was standardized, and the sensitivity and specificity of the primers were determined. nPCR technique was applied to tissue sections obtained from formalin-fixed and paraffin-embedded tissues of ERM from 23 patients with Eales' disease and 27 noninfective and non-Eales' disease patients as controls. nPCR technique was specific for M. tuberculosis genome and sensitive enough to detect 0.25 fg (corresponding to the presence of a single bacillus). Eleven (47.8%) ERM of 23 Eales' disease and 3 (11.1%) of 27 controls were positive for M. tuberculosis genome. The difference between the two groups was statistically significant (P = 0.001), indicating association of this bacterium with Eales' disease. The demonstration of the presence of M. tuberculosis DNA by nPCR technique in significant number of ERM of Eales' disease compared with the controls further emphasizes the probable role of this bacterium in the pathogenesis of this enigmatic clinical condition.
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            Novel multi-targeted polymerase chain reaction for diagnosis of presumed tubercular uveitis

            Background The objective of this study was to report the use of multi-targeted polymerase chain reaction (PCR) in the diagnosis of presumed tubercular uveitis. Multi-targeted PCR using three targets specific for Mycobacterium tuberculosis, i.e., IS6110, MPB64, and protein b, was performed on intraocular fluid samples of 25 subjects. Nine had presumed tubercular uveitis, six had intraocular inflammation secondary to a nontubercular etiology (disease controls), and ten had no evidence of intraocular inflammation (normal controls). As described previously, response to antitubercular therapy was considered as the gold standard. Results Multi-targeted PCR was positive in seven out of nine patients with presumed tubercular uveitis and negative in all normal and disease controls. The sensitivity and specificity were 77.77% and 100%, respectively. For the diagnosis of presumed tubercular uveitis, multi-targeted PCR had a positive predictive value of 100% and a negative predictive value of 88.88%. Conclusion Multi-targeted PCR can be a valuable tool for diagnosing presumed tubercular uveitis.
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              Quantitative polymerase chain reaction for Mycobacterium tuberculosis in so-called Eales' disease.

              To report mycobacterial load in the vitreous of patients labeled as having Eales' disease. Eighty-eight patients were prospectively enrolled into 3 groups: 28 patients with so-called Eales' disease (group A); 30 positive controls with specific uveitis syndromes (group B), and 30 negative controls (group C). The undiluted vitreous humor samples were collected and subjected to real-time PCR assay for MPB64 gene of Mycobacterium tuberculosis (MTB) and load quantified. Sixteen (57.14%) vitreous fluid samples in group A; 1 sample in group B, and none of the samples in group C were positive for MTB genome from the vitreous. The copies of MTB genomes in the positive samples in group A were 1.52 × 10(4) to 1.01 × 10(6). MTB genome was demonstrated in more than 50% of vitreous fluid samples with significant bacillary load, indicating that half of patients with so-called Eales' disease are indeed cases of tubercular vasculitis.
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                Author and article information

                Contributors
                +91-172-2747837 , mankoo95@yahoo.com
                Journal
                J Ophthalmic Inflamm Infect
                J Ophthalmic Inflamm Infect
                Journal of Ophthalmic Inflammation and Infection
                Springer Berlin Heidelberg (Berlin/Heidelberg )
                1869-5760
                27 February 2016
                27 February 2016
                2016
                : 6
                : 8
                Affiliations
                [ ]Advanced Eye Center, Postgraduate Institute of Medical Education and Research, Sector 12, Chandigarh, 160012 India
                [ ]Department of Medical Microbiology, Postgraduate Institute of Medical Education and Research, Chandigarh, India
                [ ]Stanley M. Truhlsen Eye Institute, University of Nebraska Medical Center, Omaha, NE USA
                [ ]Department of Internal Medicine, Postgraduate Institute of Medical Education and Research, Chandigarh, India
                Article
                74
                10.1186/s12348-016-0074-3
                4769709
                26922651
                c805022b-2560-4c2c-8f29-607ba56e9de9
                © Singh et al. 2016

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made.

                History
                : 16 December 2015
                : 22 February 2016
                Categories
                Brief Report
                Custom metadata
                © The Author(s) 2016

                Ophthalmology & Optometry
                irvan,intraocular tuberculosis,anti-tubercular therapy,aneurysms,fluorescein angiography,uveitis,polymerase chain reaction

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