Superior ophthalmic vein thrombosis (SOVT) is a rare clinical entity that may be associated with sino-orbital disease. The clinical presentation of SOVT may include signs of venous congestion, such as unilateral ptosis, chemosis, ophthalmoplegia, and eyelid swelling, with or without fundus findings. This case report describes a case of SOVT associated with orbital cellulitis diagnosed with magnetic resonance imaging and treated using anticoagulant therapy, antibiotherapy, and a corticosteroid. In the presence of orbital cellulitis, clinicians should always keep the possibility of SOVT in mind, as it may result in mortality and visual loss if not diagnosed early and given appropriate treatment without delay.