Abstract Adult Onset Still's Disease with Autoimmune Haemolytic anaemia is a rare combination, difficult to diagnose but easy to manage. Literature to date reports only two cases. Hereby, we describe first of such a case from Pakistan. A 35 year old male, known case of Autoimmune Haemolytic Anaemia, presented with the complaints of high grade fever, fatigue, myalgia, skin rash and joint pain. After thorough investigation he was diagnosed as Adult Onset Still’s Disease, according to the Yamaguchi criteria, and methylprednisolone (40 mg/day) was initiated. While his fever was relieved, dramatic improvement was seen with patient joint complaints and his anaemia was also under control. Prognosis is good with regular medication, and it is necessary to educate both patient and family, to enable them to have a complete understanding of the disease and its effects on their life. Keywords: Adult Onset Still’s disease, Autoimmune Haemolytic anaemia, AOSD, Yamaguchi criteria, Methylprednisolone, Pakistan. Introduction First described in children by George Still in 1896, Adult-onset Still's disease (AOSD) is a rare systemic 1239 J Pak Med Assoc Adult Onset Still’s Disease with Autoimmune Haemolytic anaemia: Two rarities in combination Muhammad Ishaq, 1 Jibran Sualeh Mohammad, 2 Syed Ali Haider Naqvi, 3 M. Ishaq Shaikh 4 Department of Medicine, Jinnah Medical College Hospital, 1,2,4 Sindh Govt. Hospital, Liaqatabad, 3 Karachi. Case Report