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CASE REPORT
Year :   |  Volume :   |  Issue :   |  Page :  

Rapid regression of idiopathic multifocal choroiditis with intravitreal methotrexate-wide-field optical coherence tomography angiography-based study


1 Department of Retina, Grewal Eye Institute, Chandigarh, India
2 Department of Ophthalmology, Duke University, Durham, NC, USA

Date of Submission26-Nov-2020
Date of Decision09-Jan-2022
Date of Acceptance06-May-2022
Date of Web Publication03-Aug-2022

Correspondence Address:
Manpreet Brar,
Department of Retina, Grewal Eye Institute, SCO: 168-169, Sector 9C, Chandigarh - 160 009
India
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ojo.ojo_446_20

   Abstract 


Wide-field optical coherence tomography angiography (OCTA) results in a patient of idiopathic multifocal choroiditis successfully treated with intravitreal methotrexate injections.

Keywords: Methotrexate, multifocal choroiditis, wide-field optical coherence tomography angiography



How to cite this URL:
Brar M, Singh Grewal SP, Grewal DS, Sharma M, Dogra MR. Rapid regression of idiopathic multifocal choroiditis with intravitreal methotrexate-wide-field optical coherence tomography angiography-based study. Oman J Ophthalmol [Epub ahead of print] [cited 2022 Aug 10]. Available from: https://www.ojoonline.org/preprintarticle.asp?id=353269




   Introduction Top


Systemic methotrexate (MTX) has been successfully used for ocular inflammatory disorders since 1965.[1] However, it requires 6–8 weeks for it to be completely effective;[2] thus, intravitreal (IVT) MTX was used as an off-label drug to achieve a stronger local anti-inflammatory response[3] and it also avoids systemic side effects.[4] We report multimodal imaging results including wide-field optical coherence tomography angiography (OCTA) in a steroid responder with fovea threatening idiopathic multifocal choroiditis (I-MFC) who was treated with IVT MTX.


   Case Report Top


A 45-year-old male presented with ocular pain and blurred vision in the left eye for the past 3 weeks and blurred vision in his right eye for the past 2 days. There was no history of cough, low-grade fever, skin rashes, oral/genital ulcers, skin lesions, arthritis, kidney disease, or contact with cats or dogs or relevant history of tick bites. He had been diagnosed with multifocal choroiditis in the left eye for which he had been treated with IVT triamcinolone (IVT-TCA) injection and oral corticosteroids.

On clinical examination, his best-corrected visual acuity was 20/20 in the right eye and 20/30 in the left eye. IOP measured by Goldmann Applanation Tonometry was 20 mmHg and 38 mmHg in the right eye and left eye, respectively. Slit-lamp biomicroscopy found no anterior segment inflammation, and no cells were seen in the vitreous cavity. Fundus examination of the right eye showed multiple, creamy, white, round-to-oval choroidal lesions scattered in the mid-peripheral retina and a few creamy white choroidal lesions threatening the fovea [Figure 1]a. The left eye presented with mostly healed plaque-like lesions involving the posterior pole [Figure 1]b. Wide-field OCTA montage image at the level of the choriocapillaris slab demonstrated hypo flow void spaces corresponding to active inflammatory choroidal lesions [Figure 2]. Swept source OCT scan of the right eye demonstrated hyper reflective nodularity at the level of retinal pigment epithelium (RPE) with hyper reflective subretinal lesion at the level of the outer nuclear layer with involvement of the external limiting membrane and abnormal lucency at the ellipsoid zone [Figure 3]. Basic uveitis workup including chest X-ray, Mantoux skin test, and blood test including QuantiFERON gold, angiotensin-converting enzyme, syphilis serology, hemogram, and erythrocyte sedimentation rate were all normal.
Figure 1: (a) Color fundus montage image of the right eye in a 45-year-old male with idiopathic multifocal choroiditis (acute inflammatory stage) lesions in the right eye were multifocal, creamy colored with fuzzy margins, scattered in the mid-peripheral retina and juxta foveal region. (b) Color fundus image of the left eye showed mostly healed plaque-like choroidal lesions involving the posterior pole and midperipheral retina

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Figure 2: Wide-field OCTA montage image of right eye at the level of choriocapillaris slab demonstrated round to oval hypo flow void spaces with ill-defined margins. OCTA: Optical coherence tomography angiography

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Figure 3: SS OCT scan of the right eye demonstrated hyperreflective nodularity at the level of RPE with hyperreflective subretinal lesion at the level of ONL with associated involvement of the ELM and abnormal lucency at the ellipsoid zone. SS OCT: Swept-source optical coherence tomography, RPE: Retinal pigment epithelium, ONL: Outer nuclear layer, ELM: External limiting membrane

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Systemic acetazolamide and topical (AGM) drops were started for the left eye to control the intraocular pressure. Systemic corticosteroids were tapered rapidly as the patient was a strong steroid responder.

IVT MTX (400 μg/0.1 ml) (MTX 50 mg/2 ml vial, Intas Pharmaceuticals, Ahmedabad, India) was administered in the right eye, and as a result, the choroidal lesions regressed rapidly in the midperiphery, and the juxtafoveal lesions did not show any progression at 1 week. The patient received a total of three IVT MTX injections at 1-week intervals without any adverse reaction. There was no further progression of MFC, and subsequent healing of the juxtafoveal choroidal lesions was also observed. At the last follow-up, 4 months from the baseline, vision for the right eye was stable at 20/20, and no new lesions were noticed on fundus examination [Figure 4]. Wide-field OCTA montage image showed partial restoration of vascular flow in the juxtafoveal region, and no new lesions were detected [Figure 5].
Figure 4: Color fundus montage image at 4 months showed that no new lesions were noticeable and juxta foveal scarring is seen temporal to the fovea

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Figure 5: Wide-field OCTA montage image shows abnormal vascular flow in the juxta foveal region only that has not further increased and no new flow void areas could be seen. OCTA: Optical coherence tomography angiography

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   Discussion Top


I-MFC without panuveitis or vitritis is a chronic, progressive, bilateral inflammatory chorioretinopathy with no known associated systemic or ocular diseases.[5] Principal site of involvement is RPE and outer retinal spaces. It may vary in severity from transient photoreceptor attenuation with the recovery of morphology and function to permanent CR atrophic lesions.[6] The exact mechanism involved in outer retinal dysfunction and photoreceptor death remains unclear; however, Spaide et al.[6] and others have stated that immune-modulating therapy induces recovery. In our patient, the left eye has progressed to chorioretinal scarring resulting in permanent visual disability most likely as a result of late presentation and inadequate control of the inflammatory load. However, in his right eye, where the symptoms were of a recent onset, and the disease was identified at the early stages, local IVT MTX injection was administered to achieve a quick concentrated local anti-inflammatory response in the vitreous cavity, the result was a faster regression of the disease and the fovea was spared from involvement. Ghose et al.[7] have reported similar results of quicker regression of inflammatory choroidal lesions with the use of low-dose IV TCA, and there have been a few case reports on the use of IVT corticosteroid implants for the control of persistent inflammation despite systemic immunosuppression.[8],[9] However, IOP rise is the main ocular side effect of such steroid usage.

Our case highlights the use of IVT MTX in a steroid responder as a good alternative for intensive localized anti-inflammatory effects. Use of IVT MTX in cases of MFC has been documented before in the literature, but it was mainly used as an adjunctive agent along with systemic immunosuppression in cases with persistent or refractory inflammatory activity in tubercular serpiginous such as choroiditis[9] or in cases of multifocal choroiditis associated with choroidal neovascularization.[10] Our case demonstrated quick regression and good control of inflammatory load with multiple MTX injections. Our case further highlights the use of noninvasive OCTA to diagnose and monitor the disease activity in such inflammatory ocular disorders.

Our case demonstrated that IVT MTX appears to be a promising alternative to IVTCA/corticosteroid implants, especially in steroid responders, and an extended remission effect should be explored in future studies.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient has given his consent for his/her/their images and other clinical information to be reported in the journal. The patient understands 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 Top

1.
Wong VG. Methotrexate treatment of uveal disease. Am J Med Sci 1966;251:239-41.  Back to cited text no. 1
    
2.
Shah SS, Lowder CY, Schmitt MA, Wilke WS, Kosmorsky GS, Meisler DM. Low-dose methotrexate therapy for ocular inflammatory disease. Ophthalmology 1992;99:1419-23.  Back to cited text no. 2
    
3.
Taylor SR, Habot-Wilner Z, Pacheco P, Lightman SL. Intraocular methotrexate in the treatment of uveitis and uveitic cystoid macular edema. Ophthalmology 2009;116:797-801.  Back to cited text no. 3
    
4.
Michel SS, Ekong A, Baltatzis S, Foster CS. Multifocal choroiditis and panuveitis: Immunomodulatory therapy. Ophthalmology 2002;109:378-83.  Back to cited text no. 4
    
5.
Essex RW, Wong J, Jampol LM, Dowler J, Bird AC. Idiopathic multifocal choroiditis: A comment on present and past nomenclature. Retina 2013;33:1-4.  Back to cited text no. 5
    
6.
Spaide RF, Goldberg N, Freund KB. Redefining multifocal choroiditis and panuveitis and punctate inner choroidopathy through multimodal imaging. Retina 2013;33:1315-24.  Back to cited text no. 6
    
7.
Ghose A, Bhende PS, Biswas J. Efficacy of reduced dose of intravitreal triamcinolone acetonide in a case of active serpiginous choroiditis. Indian J Ophthalmol 2016;64:681-2.  Back to cited text no. 7
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8.
Fonollosa A, Valsero S, Artaraz J, Ruiz-Arruza I. Dexamethasone intravitreal implants in the management of tubercular multifocal serpiginoid choroiditis. J Ophthalmic Inflamm Infect 2016;6:31.  Back to cited text no. 8
    
9.
Julian K, Langner-Wegscheider BJ, Haas A, De Smet MD. Intravitreal methotrexate in the management of presumed tuberculous serpiginous-like choroiditis. Retina 2013;33:1943-8.  Back to cited text no. 9
    
10.
Mateo-Montoya A, Baglivo E, de Smet MD. Intravitreal methotrexate for the treatment of choroidal neovascularization in multifocal choroiditis. Eye (Lond) 2013;27:277-8.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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