In a patient undergoing regular hemodialysis through an arteriovenous fistula access, pleural effusion is a known long term complication. However, a unilateral hemothorax is relatively uncommon. Here we report a 46 year old male, end-stage renal disease patient, on maintenance hemodialysis, who presented with a giant brachiocephalic AV fistula in his left arm and progressive breathlessness. Radiological imaging revealed a left sided pleural effusion. Ultrasound guided aspiration revealed a hemorrhagic pleural fluid. A Doppler study of the fistula revealed a high velocity blood flow through the fistula, thereby establishing the cause of the unilateral hemothorax. Ligation of the fistula resulted in complete resolution of the hemothorax. The other possible causes for hemothorax in a dialysis patient are also discussed in this case report.