Purpose: To report a rare case of ocular penicillium infection presenting as combined rhegmatogenous retinal detachment and serous retinal detachment in an immunocompetent patient. Methods: Case report.Results: A 44 year old male who had newly diagnosed type 2 diabetes mellitus. He had intermittent blurred vision in his left eye for 2 years, but progressed in recent two and half months. He had cataract surgery in the left eye 2 months before visit. Initial examination showed visual acuity was 6/60, traced cell in anterior chamber and pseudophakia. Fundus examination revealed serous retinal detachment OS first, but Shafter sign developed 11 days later. He underwent pars plana vitrectomy plus scleral buckling immediately. The pathology of vitreous biopsy demonstrated the classical picture of Penicillium marneffei. Blood culture showed no fungemia. Under the impression of ocular Penicillium infection, he received systemic intravenous infusion of Amphotericin-B 50 mg/day for 14 days and following oral Itraconazole 400 mg/day for 3 months. Besides, 0.15% Amphotericin-B eye drop was administered for next 3 week. The ocular inflammation was silent down after operation. Final visual acuity was 6/15. He didn't have any ocular recurrence or systemic manifestation during 9 months follow up.Conclusion: P. marneffei has not only emerged as an important fungal pathogen that causes disseminated infection in HIV-infected patients residing in or travelling to endemic area, but localized ocular infection of P. marneffei could also be possible in immunocompetent patients. Hence, ophthalmologists should be aware of any ocular inflammation with patient who travels to or resides in the Southeast Asia.