A case of herpes zoster uveitis with severe hyphema

Background: Uveitis sometimes causes hyphema, but severe hyphema as a complication following herpes zoster uveitis has rarely been reported. We report a rare case of zoster sine herpete with unusually severe hyphema. Case presentation: A 41-year-old Japanese female developed hyphema filling almost one-half of the depth of the anterior chamber after a two-week history of unilateral anterior uveitis. Hyphema persisted for four weeks while sectorial iris atrophy became gradually apparent. Systemic prednisolone and valaciclovir resulted in prompt resolution of uveitis and hyphema. Serum anti-varicella zoster virus (VZV) IgG measured by enzyme immunoassay was 116 at presentation and decreased to 20.3 four month later. In addition, the antibody level in aqueous humor was almost 10-fold higher than that in serum examined 9 months after presentation. Because there was no skin lesion, this case was diagnosed as zoster sine herpete. The patient underwent cataract operation due to secondary cataract. The final visual acuity in decimal notation was 1.0, but complications such as severe iris atrophy, wide anterior synechiae, corneal opacity, and decrease in corneal endothelial cell count remained. Conclusion: Zoster sine herpete is an important differential diagnosis in a case of acute anterior uveitis with severe hyphema, although such cases are quite rare. Measurement of anti-VZV IgG levels by enzyme immunoassay in aqueous humor and serum would be useful in the diagnosis of VZV reactivation. Prompt diagnosis and administration of corticosteroids and anti-herpes virus medication may improve the outcome.