RESUMEN
Ipilimumab, an immune checkpoint inhibitor used in the treatment of metastatic melanoma, can cause immune-related adverse events including rare ocular-related inflammation. This is a case of a 54-year-old man with metastatic melanoma and bilateral orbital inflammation associated with ipilimumab that occurred after drug rechallenge 6 years after initial orbital inflammation with ipilimumab use. Imaging revealed tendon-involving myositis. He was managed with intravenous corticosteroid and intravenous immunoglobulin therapy with improvement in his orbital inflammation. This case demonstrates the potential ocular effects of ipilimumab use, differentiation of ipilimumab-associated orbital inflammation from thyroid orbitopathy and management considerations to effectively reduce orbital inflammation.