This is the first report of the efficacy of leukocytapheresis (LCAP) in a patient with refractory adult onset Still's disease (AOSD) during pregnancy. A 32-year-old Chinese pregnant woman with AOSD who had been treated with prednisolone failed to achieve disease stabilization. The patient's disease was successfully controlled with the initiation of LCAP. Subsequently, she gave birth via Caesarean section to a 1,878 g baby boy at 34 weeks of gestation while maintaining remission. We conclude that LCAP is an alternative treatment in pregnant patients with refractory AOSD, particularly in those concerned about potential teratogenic and other adverse effects.