| Summary: | Purpose: To describe the clinical presentation and imaging features of a patient presenting with bilateral central retinal vein occlusion (CRVO), who was subsequently diagnosed with hyperviscosity retinopathy due to B cell lymphoproliferative disease, and had a good response to systemic immunosuppressive therapy. Observations: A clinical case report of an 87-year-old woman who presented with bilateral CRVO. Visual acuity, clinical examination, spectral domain optical coherence tomography (SD-OCT), color fundus photography and systemic evaluation were obtained. Ocular examination at presentation revealed bilateral CRVO, and OCT examination revealed significant central macular edema bilaterally. Six months after the diagnosis of hyperviscosity retinopathy and administration of systemic cyclophosphamide immunosuppressive therapy for B cell lymphoproliferative disease, most of the retinal hemorrhages resolved and partial resolution of the macular edema in the left eye was observed. Conclusion and Importance: This case describes the association between bilateral simultaneous CRVO and hyperviscosity. Under unique circumstances our patient received systemic therapy alone, without plasmapheresis. Although only limited therapy was applied, she didn't have deterioration or recurrent events; she had a small improvement in her macular edema and a significant improvement in her systemic functional state as well as reduction in her monoclonal IGM level. Keywords: Bilateral, CRVO, Hyperviscosity, Retinopathy, Waldenstrom's macroglobulinemia
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