OSLI Retina

September 2020

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532 Ophthalmic Surgery, Lasers & Imaging Retina | Healio.com/OSLIRetina lesions displayed attenuation and disruption of the el- lipsoid zone and thickening and disruption of the RPE (Figure 2F). FAF illustrated patchy hyperautofluores- cence with surrounding hypoautofluorescence of the lesions (Figure 2B). FA showed window defects in the area of the outer retinal lesions (Figure 2C), whereas ICG was normal (Figure 2D). Coxsackie titers were not performed, but a routine inflammatory and infectious work-up including HLA-B27, lysozyme, ACE, ANCA, QuantiFERON gold, antinuclear antibodies, C-reactive protein, erythrocyte sedimentation rate, RPR, and FTA was unremarkable. Observation was recommended. Upon follow-up 5 weeks later, the lesions appeared partially regressed (Figures 2G-2J). DISCUSSION Hand, foot, and mouth disease has been associated with a variety of ocular manifestations, most notably AIM. 2-5 AIM typically presents as central vision loss with an exudative maculopathy and alteration of the ellipsoid zone band with thickening of the RPE dem- onstrated by spectral domain OCT. 2,4,5,10,11 Cases of exudative maculopathy with vitritis, retinal hemor- rhages, exudation, and papillitis have been reported within the AIM spectrum. 1,5,11 AIM may present with unilateral (the most common presentation, typically called UAIM) or bilateral involvement. 2,11 The most likely cause of AIM is coxsackie virus. 3-5 This entity is generally considered to be self-limited with resolu- tion occurring within several weeks. 10,11 Upon resolu- tion, a macular scar with RPE alterations may occur, typically in a bull's-eye pattern. The development of a macular hole following AIM has also been de- scribed. 3,10,11 Both cases described in this series were associated with symptomatic hand, foot, and mouth disease, and one of the patients was positive for coxsackie serum virus titers. The cases were unilateral with well cir- cumscribed lesions occurring at the level of the el- lipsoid zone and RPE, typical of UAIM. Subretinal fluid was not observed in our cases; however, small midperipheral lesions may be less likely to develop exudative detachment compared to the large macular lesions associated with typical AIM. Haamann et al. and Freund et al. reported cases similar to our Case 1, in which a well-circumscribed area of outer reti- nitis was described. 9,11 However, Haamann et al. did not include OCT or FA during the acute phase, which is essential to fully characterize the location and ac- tivity of these lesions, and the case by Freund et al. reported negative coxsackie virus titers. Kadrmas et al. reported a case of multiple paracentral and mid- peripheral creamy chorioretinal lesions in a patient with Coxsackie virus B4 infection, 8 and these lesions resembled the solitary lesion in Case 1. The Kadrmas study was performed in the pre-OCT era, therefore one cannot be sure of the exact nature of these le- sions. Balaratnasingam et al. reported two cases of multifocal outer retinal satellite lesions associated with a central neurosensory detachment. 2 It is reason- able to conclude that there is a spectrum of coxsackie retinopathy ranging from an exudative maculopathy versus multifocal disease complicated by peripapil- lary or peripheral retinal lesions. It is important to consider coxsackie virus infec- tion in the differential diagnosis of extramacular le- sions with outer retinal disruption. The term "uni- lateral acute idiopathic maculopathy" should be replaced with "coxsackie retinopathy" to better align with the expanded clinical spectrum and the known etiology of this disorder. REFERENCES 1. Tandon M, Gupta A, Singh P, Subathra GN. Unilateral hemorrhagic maculopathy: an uncommon manifestation of hand, foot, and mouth disease. Indian J Ophthalmol. 2016;64(10):772-774. https://doi. org/10.4103/0301-4738.195014 PMID:27905343 2. Balaratnasingam C, Lally DR, Tawse KL, et al. A unique posterior segment phenotypic manifestation of coxsackie virus infection. Re- tin Cases Brief Rep. 2016;10(3):278-282. https://doi.org/10.1097/ ICB.0000000000000250 PMID:26584330 3. Ghazi NG, Daccache A, Conway BP. Acute idiopathic maculopathy: report of a bilateral case manifesting a macular hole. Ophthalmology. 2007;114(5):e1-e6. https://doi.org/10.1016/j.ophtha.2006.08.055 PMID:17258809 4. Meyerle CB, Yannuzzi LA. Acute positive titers of antibody to coxsackievirus in acute idiopathic maculopathy. Retin Cas- es Brief Rep. 2008;2(1):34-35. https://doi.org/10.1097/01. iae.0000243065.91330.e0 PMID:25389612 5. Beck AP, Jampol LM, Glaser DA, Glasser DA, Pollack JS. Is cox- sackievirus the cause of unilateral acute idiopathic maculopathy? Arch Ophthalmol. 2004;122(1):121-123. https://doi.org/10.1001/ archopht.122.1.121 PMID: 14718310 6. Mine I, Taguchi M, Sakurai Y, Takeuchi M. Bilateral idiopathic retinal vasculitis following coxsackievirus A4 infection: a case report. BMC Ophthalmol. 2017;17(1):128. https://doi.org/10.1186/s12886-017- 0523-2 PMID:28724375 7. Förster W, Bialasiewicz AA, Busse H. Coxsackievirus B3-associated panuveitis. Br J Ophthalmol. 1993;77(3):182-183. https://doi. org/10.1136/bjo.77.3.182 PMID:8384474 8. Kadrmas EF, Buzney SM. Coxsackievirus B4 as a cause of adult cho- rioretinitis. Am J Ophthalmol. 1999;127(3):347-349. https://doi. org/10.1016/S0002-9394(98)00322-5 PMID:10088751 9. Haamann P, Kessel L, Larsen M. Monofocal outer retinitis associated with hand, foot, and mouth disease caused by coxsackievirus. Am J Ophthalmol. 2000;129(4):552-553. https://doi.org/10.1016/S0002- 9394(99)00440-7 PMID:10764878 10. Yannuzzi LA, Jampol LM, Rabb MF, Sorenson JA, Beyrer C, Wil- cox LM. Unilateral acute idiopathic maculopathy. Arch Oph- thalmol. 1991;109(10):1411-1416. https://doi.org/10.1001/ar- chopht.1991.01080100091049 PMID: 1929931 11. Freund KB, Yannuzzi LA, Barile GR, Spaide RF, Milewski SA, Guyer DR. The expanding clinical spectrum of unilateral acute idiopathic maculopathy. Arch Ophthalmol. 1996;114(5):555-559. https://doi. org/10.1001/archopht.1996.01100130547007 PMID: 8619764

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