Purpose: To describe a case of presumed ocular toxoplasmosis.Method: A retrospective chart review.Result: This case report describes a 35-year-old male who presented hypopyon anterior uveitis in his left eye. Fundus examination revealed severe vitritis which precluded the view of retina. His serum anti-toxoplasma immunoglobulin (Ig)G was significantly raised, with a normal anti-toxoplasma IgM level and PCR from nested primers targeting B1 gene of Toxoplasma gondii was positive. He was started on empirical anti-toxoplasma therapy. Undiluted vitreous specimen collected during lensectomy and diagnostic vitrectomy in left eye was also positive for nested primers targeting B1 gene of T. gondii.Conclusion: Ocular toxoplasmosis without retinochoroiditis, though extremely rare, can occur.