Background: Isolated ocular relapse of acute lymphoblastic leukemia is not commonly seen. Subretinal infiltration and exudative retinal detachment may not provide a positive vitreous biopsy for histopathologic confirmation.
Method: We present a case of a 5-year-old male patient with acute lymphoblastic leukemia in remission, who presented with bilateral subretinal leukemic infiltrates and exudative retinal detachment. An ocular biopsy through external needle drainage histopathologically confirmed an ocular relapse of acute lymphoblastic leukemia.
Results: Isolated ophthalmic relapse of acute lymphoblastic leukemia can present as bilateral exudative retinal detachment with subretinal infiltration.
Conclusion: External needle drainage of the subretinal fluid and subretinal infiltration can increase the diagnostic yield for histopathologic confirmation of the relapse.