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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 2  |  Issue : 2  |  Page : 424-426

A pediatric case of West Nile virus chorioretinitis associated with unilateral acute idiopathic maculopathy


1 Department of Ophthalmology, Fattouma Bourguiba University Hospital, Faculty of Medicine, University of Monastir, Monastir, Tunisia
2 Department of Pediatrics, Fattouma Bourguiba University Hospital, Faculty of Medicine, University of Monastir, Monastir, Tunisia

Date of Submission07-Oct-2021
Date of Acceptance14-Dec-2021
Date of Web Publication13-Apr-2022

Correspondence Address:
Moncef Khairallah
Department of Ophthalmology, Fattouma Bourguiba University Hospital, 5019, Monastir
Tunisia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_2577_21

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  Abstract 


We report a pediatric case of unilateral acute idiopathic maculopathy (UAIM) associated with West Nile virus (WNV) infection. A 10-year-old child with a 15-day history of presumed acute viral encephalitis, complained of blurred vision in the right eye. Clinical and multimodal imaging findings, including disruption of the foveal ellipsoid zone (EZ) with preservation of the external limiting membrane on Swept Source OCT (SS OCT), were consistent with UAIM. The finding of associated curvilinear chorioretinal lesions in the setting of encephalitis led a diagnosis of WNV infection to be considered and subsequently confirmed by serology. The EZ spontaneously restored over a few weeks with near complete visual recovery. This is a unique pediatric case of UAIM associated with serologically proven WNV infection.

Keywords: Multifocal chorioretinitis, optical coherence tomography, optical coherence tomography angiography, unilateral acute idiopathic maculopathy, West Nile virus


How to cite this article:
Ben Amor H, Daoud M, Besbes H, Ksiaa I, Chouchane S, Jelliti B, Khochtali S, Khairallah M. A pediatric case of West Nile virus chorioretinitis associated with unilateral acute idiopathic maculopathy. Indian J Ophthalmol Case Rep 2022;2:424-6

How to cite this URL:
Ben Amor H, Daoud M, Besbes H, Ksiaa I, Chouchane S, Jelliti B, Khochtali S, Khairallah M. A pediatric case of West Nile virus chorioretinitis associated with unilateral acute idiopathic maculopathy. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 May 27];2:424-6. Available from: https://www.ijoreports.in/text.asp?2022/2/2/424/342968



Multifocal chorioretinitis (CR), usually associated with neurological disease, advanced age, and diabetes, is the most common ocular manifestation of West Nile virus (WNV) infection. Most reported patients with WNV CR were over 50 years in age, and pediatric cases are very rare. Chorioretinal lesions arranged in a scattered or more typically linear pattern can involve the midperiphery or posterior pole.[1]

The foveal center is usually spared, but central vision loss can rarely occur due to occlusive retinal vasculitis, macular edema, or choroidal neovascularization.[2],[3]

We herein report a pediatric case of WNV-associated UAIM, documented with multimodal imaging, including Swept Source OCT (SS OCT) and OCT Angiography (OCTA).


  Case Report Top


A 10-year-old child was referred for evaluation of rapid decreased vision in his right eye (RE). He had been admitted 15 days earlier to the pediatric department for presumed acute viral encephalitis. He denied pain, photophobia, flashes, or floaters. His best-corrected acuity was 20/50 in the RE and 20/20 in the left eye (LE). Intraocular pressure was normal in both eyes. There were no anterior segment or vitreous cells in either eye. Fundus examination of the RE showed a yellowish-grey subfoveal lesion with indistinct borders and a rough bull's-eye appearance. A curvilinear streak of discrete, flat, whitish-yellow, deep retinal lesions extended from the temporal margin of the optic disc to the subfoveal lesion [Figure 1]a. There was no evidence of retinal vasculitis, papillitis, or other fundus abnormalities in the RE. The LE posterior segment was unremarkable.
Figure 1: (a) Fundus photograph of the right eye showing a bull's-eye-shaped yellowish-grey discoloration of the central macula (arrow). Numerous deep, whitish-yellow retinal lesions extend in a curvilinear pattern from the temporal margin of the optic disc to the subfoveal lesion (arrowhead). (b) Late-phase fluorescein angiogram showing central hypofluorescence surrounded by speckled hyperfluorescence (arrow) and faint hyperfluorescence of associated chorioretinal lesions (arrowhead). (c) Fundus autofluorescence showing central hyperautofluorescence with surrounding hypoautofluorescence and well-delineated, uniformly hypoautofluorescent spots extending from the optic disc to the subfoveal lesion. (d) SS OCT of the macula showing disruption and irregularity of the ellipsoid zone with preservation of the external limiting membrane and hyperreflective material on the apical side of the retinal pigment epithelium

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Fluorescein angiography of the RE showed early central hypofluorescence with surrounding faint, ring-shaped hyperfluorescence corresponding to the subfoveal lesion seen clinically. It also demonstrated hyperfluorescence of the interpapillo-macular curvilinear chorioretinal lesions [Figure 1]b. Fundus autofluorescence (FAF) showed central hyperautofluorescence with surrounding hypoautofluorescence and multiple well-delineated, uniformly hypoautofluorescent spots extending from the optic disc to the subfoveal lesion [Figure 1]c. SS OCT of the macula revealed disruption and irregularity of the ellipsoid zone (EZ) with preservation of the external limiting membrane and hyperreflective material on the apical side of the retinal pigment epithelium (RPE), inducing irregular thickening [Figure 1]d. The subfoveal choroidal thickness was 330 μm. All imaging findings in the LE were unremarkable, and the subfoveal choroidal thickness was 294 μm.

Results of laboratory investigations including syphilis serology, Chest X-ray, and tuberculosis test were unremarkable.

The patient initially was diagnosed with unilateral acute idiopathic maculopathy (UAIM). The finding of associated curvilinear chorioretinal lesions in the setting of encephalitis led us to consider a diagnosis of WNV infection. This was confirmed by detection of West Nile–specific IgM in serum by an enzyme-linked immunosorbent assay (ELISA).

Three weeks after initial presentation, visual acuity had improved to 20/25. On fundus examination, there was a persistent “bull's-eye” pattern, and the curvilinear lesions became more evident [Figure 2]a. The FAF findings remained unchanged. SS OCT showed a near-complete restoration of the EZ with a residual slight RPE elevation [Figure 2]b. The subfoveal choroidal thickness was 299 μm. SS OCTA showed an area of signal loss at the level of the choriocapillaris corresponding to the foveal bull's-eye lesion. There were also small areas of signal loss corresponding to the associated curvilinear chorioretinal lesions [Figure 2]c and [Figure 2]d. Over the following 3 months, visual acuity remained stable, and sequential multimodal imaging showed no further significant changes.
Figure 2: (a) Fundus photograph taken 3 weeks after initial presentation showing a persistent central “bull's-eye” pattern with associated inactive chorioretinal lesions. (b) SS OCT showing a near-complete restoration of the ellipsoid zone with a residual slight nodular retinal pigment epithelium elevation. (c) SS OCTA showing an area of signal loss at the level of the choriocapillaris corresponding to the area of bull's-eye lesion and small areas of signal loss corresponding to the associated curvilinear chorioretinal lesions. (d) B-scan OCTA showing decreased flow signal in the choriocapillaris that does not seem to be related to shadowing

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  Discussion Top


This child was initially diagnosed as having UAIM based on typical clinical and imaging findings, and spontaneous favorable course.[4],[5],[6] The finding of associated linearly arranged chorioretinal lesions in the setting of febrile encephalitis led a diagnosis of WNV infection to be considered and subsequently confirmed with serology.[1]

Unilateral acute idiopathic maculopathy has been previously associated with Coxsackievirus infection, acquired Zika, and yellow fever.[7],[8],[9] Our case shows that WNV, like these viruses, can induce acute maculopathy consistent with UAIM. It is unclear whether macular disease results from a direct effect of the virus itself or from an immune mediated reaction.

Our findings, consistent with previous data,[8],[9],[10] show that multimodal imaging is useful in confirming the diagnosis of UAIM, and in monitoring the disease course. Multimodal imaging in our patient also was helpful in detecting and characterizing associated multifocal choroiditis. SS OCT was especially valuable in documenting the disruption of the foveal EZ, which was correlated with visual loss. On the other hand, the near-complete restoration of the EZ during the convalescent phase of the disease corresponded to visual recovery.

It is widely thought that the pathogenesis of UAIM involves inflammation of the inner choroid, retinal pigment epithelium, and outer retina that is partially reversible.[8],[10] Recent SS OCTA data showed evidence of a partially reversible choriocapillaris flow reduction in a patient with UAIM.[11] In our patient, an area of choriocapillaris flow deficit was still visible after restoration of the EZ, and it was associated with multifocal hypointense areas, corresponding to atrophic chorioretinal lesions. The OCTA data suggest that a primary or secondary choriocapillaritis with associated reversible outer retinal alterations may be involved in the pathogenesis of UAIM.


  Conclusion Top


Our case shows that West Nile virus infection should be considered in patients with UAIM from geographic areas where the virus is endemic. A careful analysis of clinical and multimodal imaging findings is essential not to miss associated typical chorioretinitis.

Acknowledgements

This work has been supported by the Ministry of Higher Education and Research of Tunisia.

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 Top

1.
Khairallah M, Ben Yahia S, Ladjimi A, Zeghidi H, Ben Romdhane F, Besbes L, et al. Chorioretinal involvement in patients with West Nile virus infection. Ophthalmology 2004;111:2065-70.  Back to cited text no. 1
    
2.
Khairallah M, Ben Yahia S, Attia S, Jelliti B, Zaouali S, Ladjimi A. Severe ischemic maculopathy in a patient with West Nile virus infection. Ophthalmic Surg Lasers Imaging 2006;37:240-2.  Back to cited text no. 2
    
3.
Chan CK, Limstrom SA, Tarasewicz DG, Lin SG. Ocular features of West Nile virus infection in North America: A study of 14 eyes. Ophthalmology 2006;113:1539-46.  Back to cited text no. 3
    
4.
Lindsey NP, Hayes EB, Staples JE, Fischer M. West Nile virus disease in children, United States, 1999–2007. Pediatrics 2009;123:e1084-9.  Back to cited text no. 4
    
5.
Messacar K, Cree-Green M, Lovell M, Anderson MS, Dominguez SR. Severe neuroinvasive West Nile virus infection in a child with undiagnosed Addison's disease. IDCases 2014;1:29-31.  Back to cited text no. 5
    
6.
Beck AP, Jampol LM, Glaser DA, Pollack JS. Is coxsackievirus the cause of unilateral acute idiopathic maculopathy? Arch Ophthalmol 2004;122:121-3.  Back to cited text no. 6
    
7.
Yannuzzi LA, Jampol LM, Rabb MF, Sorenson JA, et al. Unilateral acute idiopathic maculopathy. Arch Ophthalmol 1991;109:1411-6.  Back to cited text no. 7
    
8.
Jung CS, Payne JF, Bergstrom CS, Cribbs BE, Yan J, Hubbard GB 3rd, et al. Multimodality diagnostic imaging in unilateral acute idiopathic maculopathy. Arch Ophthalmol 2012;130:50-6.  Back to cited text no. 8
    
9.
Srour M, Querques G, Rostaqui O, Souied EH. Early spectral-domain optical coherence tomography findings in unilateral acute idiopathic maculopathy. Retina 2013;33:2182-4.  Back to cited text no. 9
    
10.
Haruta H, Sawa M, Saishin Y, Ohguro N, Tano Y. Clinical findings in unilateral acute idiopathic maculopathy. Int Ophthalmol 2010;30:199-202.  Back to cited text no. 10
    
11.
Nicolo M, Rosa R, Musetti D, Musolino M, Traverso CE. Early swept-source optical coherence tomography angiography findings in unilateral acute idiopathic maculopathy. Ophthalmic Surg Lasers Imaging Retina 2016;47:180-2.  Back to cited text no. 11
    


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