Ocular involvement in non-Hodgkin lymphoma occurs either as primary ocular, central nervous system lymphoma or isolated intraocular lymphoma. Recurrence of systemic non-Hodgkin lymphoma in the form of CNS lymphoma has been reported. However, recurrence as an isolated optic nerve lesion without involving CNS has never been reported in the pediatric age group. We report a case of systemic diffuse large B-cell lymphoma in a 2-year-old female, which primarily occurred as suprapubic mass and later recurred in the form of isolated optic nerve infiltration, after remission of the primary disease. Early detection and prompt treatment resulted in complete reversal of the disease.