Abstract
PURPOSE: This report presents an unusual case of bilateral diffuse uveal melanocytic proliferation (BDUMP) associated with a macular hole that resolved after choroidal thinning following intravitreal methotrexate and systemic chemotherapy. CASE REPORT: A 78-year-old male presented with progressive visual deterioration, significant weight loss and dyspnea. Ophthalmologic examination revealed multiple elevated yellowish-white lesions and few vitreous cells in both eyes, with a macular hole in the left eye. Optical coherence tomography showed subretinal fluid, hyperreflective lesions and choroidal irregularities at posterior pole in both eyes. Systemic screening revealed a pulmonary nodule, prompting further investigation. A pleural biopsy disclosed diffuse infiltration by mature-looking lymphocytes, positive for CD20 and BCL-2, indicating a marginal zone B-cell lymphoma. The patient received systemic chemotherapy and off-label intravitreal methotrexate injections. Notably, the macular hole in the left eye spontaneously closed following treatment, leading to significant visual improvement. DISCUSSION: BDUMP poses diagnostic and therapeutic challenges necessitating a multidisciplinary approach. This case is notable for the mechanism of macular hole closure after intravitreal methotrexate and choroidal thinning. We believe that systemic chemotherapy also played an effective role in ocular symptoms management.