Pleural involvement as one of first manifestations of a hematolymphoid malignancy is conventionally associated with solid tumors such as carcinomas of the breast, lung, gastrointestinal tract and lymphomas. A leukemic involvement of pleural effusion has seldom been described. We report a case of a 68 year old male who presented with a pleural leukemic infiltration. The patient complained of progressive exertional dyspnea for two weeks with cough and hemoptysis. The chest radiograph showed a bilateral pleural effusion. Diagnostic thoracocentesis suggested an exudate; with cytology remarkable for leukemic blasts comprising a total of 56%. The subsequent peripheral smear and bone marrow examination confirmed an acute myeloid leukemia (AML-M4). Chemotherapy was initiated but the patient succumbed shortly. This case is unique as it highlights the unusual presentation of the disease and also demonstrates the importance of analysis of material obtained in the body fluid.