• Purpose: To demonstrate the effect of oral prednisone in the treatment of atypical isolated central lesions in two patients with serpiginous choroiditis. • Methods: Two patients with a history of serpiginous choroiditis in the fellow eye developed an isolated central lesion. One patient showed combined detachment of the retinal pigment epithelium and serous detachment of the retina, while the other showed thickening of the retinal pigment epithelium and shallow detachment of the retina. Both patients were treated with 80 mg/day of oral prednisone. • Results: In both patients regression of the lesion and improvement of visual acuity was achieved within several days with- out the development of a chorioretinal scar. In one patient the le- sion recurred after discontinuation of the prednisone. With continuation of a low dose of prednisone for sev- eral months definite regression of the esion and improvement of visual acuity to near-normal values was achieved in both patients. • Conclusion: In two patients with visual loss due to an atypical central lesion in serpiginous choroiditis, regression of the lesion without the development of a typical chorioretinal scar and subsequent improvement of visual acuity was achieved with the administration of oral prednisone.