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Jay Arora, Douglas R Lazzaro, Roman Shinder, Allison E. Rizzuti; Chronic Hypertrophic Herpes Simplex Virus Infection of the Eye Masquerading as IgG4-related Disease. Invest. Ophthalmol. Vis. Sci. 2018;59(9):3649.
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To report the first case of chronic hypertrophic herpes simplex virus (HSV) infection of the eyelid and cornea masquerading as IgG4-related disease and resulting in severe vision loss.
A 37-year old African American female with a past medical history of human immunodeficiency virus (HIV) on highly active antiretroviral therapy (HAART), presented with an ulcerative lesion of the left upper and lower eyelids, and severe conjunctival inflammation with symblepharon (fig 1a). Initially, eyelid biopsy revealed findings consistent with IgG4-related disease (Fig 1 b,c), and the patient was treated with high dose oral prednisone. After one week of therapy, there was no improvement in the patient’s condition and she developed a corneal epithelial defect which progressed to chronic ulceration. Repeat biopsy and corneal cultures revealed HSV type 2 (fig 2 a,b). The patient was treated with high dose acyclovir, and the lid lesion improved. The conjunctival inflammation and corneal epithelial defect resolved but she ultimately developed corneal vascularization and opacification resulting in visual acuity of hand motions.
Chronic hypertrophic herpes simplex virus infection is a rare manifestation of HSV type 2 that affects patients with HIV. The vast majority of cases of hypetrtrophic HSV affect the genital area, and are often difficult to manage due to misleading biopsy results and poor response to standard antiviral therapy. While there are very few reported cases of hypertrohpic HSV affecting the eyelid, this is the first case of hypertrophic HSV to also affect the globe, resulting in corneal ulceration and severe vision loss.
This is an abstract that was submitted for the 2018 ARVO Annual Meeting, held in Honolulu, Hawaii, April 29 - May 3, 2018.
Fig 1: a) Ulcerative lesion of the upper and lower eyelids. b) Histopathology revealing a dense polytypic plasma cell infiltrate, fibrosis, and phlebitis. c) Greater than 100 IgG4-positive plasma cells per high power field with IgG4/IgG ratio > 40% consistent with IgG-4 related disease.
Fig 2: a) Syncytial and scattered epithelial cells with a viral cytopathic effect including smudged, "ground glass" intranuclear inclusions with acute inflammation and necrosis. b) HSV immunostain reveals scattered HSV positive cells.
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