A 35-year-old man had splenic rupture just after starting antimalarial treatment with atovaquone-proguanil followed by artesunate for acute Plasmodium ovale sensu lato infection. Spleen histology showed a 10-fold accumulation of intact P. ovale s.l.-infected erythrocytes in the spleen parenchyma compared to the general circulation. Infected erythrocytes also accumulated in small extra-splenic blood vessels, suggesting cytoadherence.