|Year : 2022 | Volume
| Issue : 2 | Page : 537-539
Paradoxical reaction presenting with orbital signs in possible intraocular tuberculosis: A case report
Suganeswari Ganesan1, Arkaprava Pradhan1, Nataraj Palaniappan2, Neethu Latiff1, Muna Bhende1, Sudha K Ganesh1
1 Shri Bhagwan Mahavir Vitreoretinal Services, Sankara Nethralaya, Chennai, Tamil Nadu, India
2 Rainbow Children Hospital, Chennai, Tamil Nadu, India
|Date of Submission||30-Jun-2021|
|Date of Acceptance||16-Oct-2021|
|Date of Web Publication||13-Apr-2022|
Shri Bhagwan Mahavir Vitreoretinal Services, Medical Reasearch Foundation, Sankara Nethralaya, 18, College Road, Nungambakkam, Chennai, Tamil Nadu - 600 006
Source of Support: None, Conflict of Interest: None
We report an unusual presentation of paradoxical reaction (PR) with orbital signs in a case of possible intraocular tuberculosis (POTB). A 15-year-old boy presented with vitreous hemorrhage in the right eye (OD) and multifocal choroiditis in the left eye (OS) and progressed to develop bilateral proptosis, lid edema, chemosis, subretinal lesions, and progression of choroidal lesions following the rapid tapering of oral steroids with anti-tubercular therapy for POTB. The orbital reaction and progressing posterior segment lesions were managed with pulse steroid therapy, oral steroids, intravitreal anti-vascular endothelial growth factor (anti-VEGF), and steroid implant. The orbital involvement is rarely seen in POTB with PR.
Keywords: Ocular tuberculosis, orbital signs, paradoxical reaction
|How to cite this article:|
Ganesan S, Pradhan A, Palaniappan N, Latiff N, Bhende M, Ganesh SK. Paradoxical reaction presenting with orbital signs in possible intraocular tuberculosis: A case report. Indian J Ophthalmol Case Rep 2022;2:537-9
|How to cite this URL:|
Ganesan S, Pradhan A, Palaniappan N, Latiff N, Bhende M, Ganesh SK. Paradoxical reaction presenting with orbital signs in possible intraocular tuberculosis: A case report. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 May 27];2:537-9. Available from: https://www.ijoreports.in/text.asp?2022/2/2/537/342896
Paradoxical reaction (PR) to anti-tubercular therapy is believed to be mediated by the host's immune system due to an enhanced delayed hypersensitivity and decreased suppressor mechanisms. It may occur in approximately 14% of the patients within 6 weeks of the initiation of therapy. The manifestations are anterior uveitis, retinal vasculitis, multifocal choroiditis, and pan-uveitis. It is essential to identify this paradoxical worsening in ocular TB to prevent sight-threatening manifestations of the disease.
| Case Report|| |
A 15 year-old immunocompetent male presented with defective vision in both eyes associated with floaters in the right eye for the past 1 month. He had undergone sling surgery for the correction of congenital ptosis 5 years back. His bestcorrected visual acuity (BCVA) was 20/40 in both eyes. The slit-lamp bio-microscopy was unremarkable with vitreous cells in the anterior vitreous. The fundus examination and ultrasound examination (USG) showed vitreous hemorrhage in OD [Figure 1]a and multifocal choroiditis in OS [Figure 1]b. Swept-source optical coherence tomography (SSOCT) showed thickened Retinal pigment epithelium (RPE) and choroid in OS [Figure 1]c. The investigations revealed a positive tuberculin skin test (TST) and a mildly enlarged hilar lymph node on the High resolution computed tomography (HRCT) chest. The polymerase chain reaction (PCR) from aqueous aspirate was negative for Mycobacterium tuberculosis TB) (MPB64 genome). The patient was started on four drugs firstline Antitubercular treatment (ATT) along with systemic steroids (1 mg/kg body weight). But the systemic steroids were rapidly tapered as there was a suspicion of central serous chorioretinopathy in OS. Within 3 days of tapering of the steroids, he presented with severe orbital pain, proptosis, lid edema, conjunctival congestion, and chemosis [Figure 2]a. The BCVA of 20/40 in OD and a counting finger in the OS were recorded. The fundus examination showed persistent vitreous hemorrhage in OD and progressive multifocal choroiditis, increasing Subretinal fluid (SRF), and subretinal hypopyon in OS [Figure 2]b and [Figure 2]c. We investigated with USG [Figure 2]d, MRI [Figure 3]a, SSOCT [Figure 3]b, and FFA [Figure 3]c and [Figure 3]d. Three doses of 1 g intravenous methylprednisolone along with ATT followed by oral steroid were given. His BCVA was counting finger and 20/400 in OD and OS, respectively. Vitrectomy was decided as he had a persistent vitreous hemorrhage and he regained good vision in OD after vitrectomy with an anti-VEGF injection. The PCR from the vitreous was positive for toxoplasma and negative for MTB (IS6110 and MPB64). Anti-toxoplasma therapy with trimethoprim and sulfamethoxazole was given for 4 weeks along with ATT and corticosteroids. During the follow-up visit, the fundus showed regressed lesions in OD while the OS showed worsening which was treated with intravitreal anti-VEGF followed by a dexamethasone implant (DI). Topical antiglaucoma medication was given in both eyes for IOP control. Anti-VEGF was repeated for cystoid macular edema (CME) [Figure 4]c in OD. At 1 year after completion of ATT, he maintained BCVA of 20/25 and 20/80 in OD and OS, respectively, with a stable fundus [Figure 4]a,[Figure 4]b,[Figure 4]c,[Figure 4]d,[Figure 4]e,[Figure 4]f.
|Figure 1: (a) Ultrasound-B scan of the right eye, shows moderate dot echoes in vitreous suggestive of vitreous hemorrhage, (b) Fundus photo of the left eye shows creamy white lesion temporal to fovea suggestive of active choroiditis, (c) Swept-source OCT of the left eye shows thickened hyper-reflectivity in RPE|
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|Figure 2: (a) External photo shows bilateral chemosis, periorbital edema, (b) Left eye Optos shows the extent of active choroiditis, (c) Fundus photo shows active choroiditis at the macula, (c) left eye colour fundus photo shows active choroiditis lesions, (d)-USG B scan of the left eye shows choroidal and scleral thickening|
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|Figure 3: (a) MRI (T2) shows the hypointense area in the sclera due to thickened scleral coat, (b) SSOCT of the left eye shows dome-shaped retinal elevation, bacillary detachment, (c) Early phase FFA of the left eye shows leakage surrounded by hypofluorescent area due to subretinal hypopyon, (d) Late phase FFA shows pooling and leakage from the vessel wall at ITQ|
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|Figure 4: (a) Fundus photo of the right eye shows healed choroidal lesion temporally; b-Fundus photo of the left eye shows healed choroidal lesion at the posterior pole; (c, e) Follow-up swept-source OCT scan of the right eye shows bumpy RPE and ellipsoid layer defects, Intra-retinal cystic spaces which resolve later; (d, f) Follow-up swept-source OCT scan of the left eye shows normal foveal contour with thickened, distorted outer retinal layers, RPE, and choroid at the macular region suggestive of healed serpiginous choroiditis|
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| Discussion|| |
This case report shows severe orbital and progressive ocular inflammation, following the initiation of ATT and steroids in a POTB which is clinically resembling central serous chorioretinopathy (CSCR), so the steroid was tapered rapidly. But later, the absence of leak in the early phase of FFA and presence of multiple lobular hyper-fluorescence with perivascular staining presenting in the mid-Arterio venous (AV) phase was not in favor of CSCR but was suggestive of PR. PR occurs in immunocompetent individuals whose bodies can incite a massive inflammatory response against infectious agents like tuberculous, nontuberculous mycobacteria, Treponema pallidum, and various viruses. Vulnerability to PR is high among the adolescent age group. Ruling out orbital cellulitis poses a major challenge when a patient with previous frontalis sling surgery presents with orbital signs. The MRI of the orbit was done to exclude orbital cellulitis. To date, the orbital signs as one of the presentations in PR have never been reported. The varied presentations of PR are the occurrence of new lesions at the same or different sites and the worsening of the existing lesions., The treatment involves escalating doses of the steroid and immune-suppressants. Identification of a PR is crucial as one needs to differentiate multidrug resistance TB from poor compliance to therapy to non-tubercular choroiditis. Serial SSOCT is helpful in monitoring the response to combination treatment (pulse steroid therapy, oral steroid, ATT, and anti-VEGF). The progression of a serpiginous choroidal granulomatous lesion showed up as increasing choroidal thickness, the dome-shaped elevation of the neurosensory retina, increasing SRF, hyper-reflectivity in the outer retina, and accumulation of subretinal hype-reflective material. Regression is noted as consolidation of choroidal granuloma and drying up of SRF. The described OCT features are hyper-reflectivity in the outer retina, loss of inner and outer segment junction, loss of RPE, and early choroidal hyper-reflectivity called the “waterfall” effect. The terminology of POTB was first alluded to by Gupta et al. The presence of bilateral multifocal serpiginous choroiditis, choroidal granuloma, and positive Mantoux test, positive findings on HRCT chest with negative microbiological evidence of TB infection from intraocular fluids made us categorize our patient as possible intraocular TB. The paucibacillary nature of the disease or presence of low copies of the MTB isolates or 2 months post-ATT collection of vitreous was the suspected reason for negative PCR. The usage of adjunct anti-VEGF in PR to achieve effective drying up of SRF, with regression of granuloma and serpiginous choroiditis, has been described by Jain et al. Ranibizumab was used for the regression of the granuloma and CME. Dexamethasone implant (DI) provided a prolonged and sustained intraocular corticosteroid delivery which was used to stabilize the OS. The safety of DI in 17 patients with tubercular uveitis was reported by Aniruddh et al.
| Conclusion|| |
Early identification of PR and prompt management with a combination therapy like intravenous steroid, ATT, oral steroids, and anti-VEGF yields good outcomes when the orbital and posterior segment involvement occurs in POTB.
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.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4]