Up to date, symptomatic neonate who needed treatments due to mother's AOSD has not been reported. We experienced a neonate born of mother diagnosed with AOSD a week before delivery. Despite treatment, the mother's symptoms had not been improved until delivery. Our patient was affected by her mother's condition, and exhibited several symptoms (fever over one week, poor activity, poor feeding) since 5 days of age, with thrombocytopenia, negative results on viral and bacterial studies, and elevation of CRP, OT/PT and ferritin. Treatment with steroid, NSAID and IV immunoglobulin was started on 14 days of age under the impression of possible maternal effects from uncontrolled AOSD of mother. The symptoms were gradually improved. Ferritin level was decreased, others were normal on 29 days of age. We present this case for the first time as effects of maternal AOSD on newborn, with review of literature.