AIM:To report a rare case of acute onset of proptosis secondary to subcutaneous panniculitis-like T cell lymphoma (SPTCL).METHODS:A case report.RESULTS:A 27-year-old Malay man presented with history of acute onset of proptosis in the left eye for 2 weeks.It was associated with history of prolonged high fever.He also developed multiple erythematous swelling on his body and both thighs during this period.On examination,visual acuity in both eyes was 6/6.The left eye was proptosed and chemotic.The extraocular move-ment was limited in all directions.The cornea and the anterior segment were normal.Fundoscopy showed normal optic disc and retina.The examination of the right eye was unremarkable.His body temperature was 40.0℃.There was presence of multiple erythematous subcu-taneous lesions over the body mainly in the left axillary,right hypochondriac,both thighs and the suprapubic area.The inguinal lympnodes were also palpable.MRI of orbit and brain revealed generalized soft tissue swelling in the left extraconal and retro-orbital space suggesting of inflammatory reaction.The cavernous sinus and brain were normal.Skin biopsy from the erythematous lesion revealed as SPTCL.He was referred to the haematologist and was started on chemotherapy-CHOP regime.The patient responded well to the chemotherapy and the proptosis reduced.CONCLUSION:Proptosis secondary to SPTCL is very rare.This is a variant of a peripheral T cell lymphoma characterised by multiple subcutaneous nodules presented with proptosis and fever.