Kyrieleis like plaques - atypical presentation of ocular Behcet's disease

Purpose: To report an unusual presentation with kyrieleis like plaques in a patient with ocular Behcet's disease. Case presentation: A 28-year-old young male presented with blurring of vision in the left eye, fundus examination revealing focal segmental intra-arterial plaques involving all branches of retinal artery characteristics of kyrieleis-like plaques with no features of retinitis in retinal periphery and mild vitritis. All routine investigations were normal, the patient testing positive for HLA-B51 marker, diagnosed as a case of ocular Bechet's disease and managed with oral steroids and immunosuppressive agents. Results: The patient had good recovery of vision with substantial resolution of intra-arterial plaques. Conclusion: Ocular Bechet's disease can present with uncommon features of kyrieleis like plaques. Ophthalmologists need to be vigilant for the presence of such potential ocular manifestations as the likely initial presentation in order to obtain early diagnosis and initiate timely management.