Both anemia and sleep disordered breathing are common in patients with dialysis-dependent stage 5 chronic kidney disease. Erythrocytosis resulting from obstructive sleep apnea (OSA) is rare in the general population and has never been described in the hemodialysis population. We present a case of asymptomatic isolated erythrocytosis and elevated serum erythropoietin level in an otherwise well and previously erythropoietin-dependent chronic hemodialysis patient with chronic kidney disease secondary to ischemic nephropathy. There was no history or symptoms of cardio-pulmonary or hepatic diseases nor any relevant family history. Screening work-up for malignancies was negative. The clinical history was highly suggestive of OSA and severe OSA (respiratory disturbance index of 59) was confirmed by polysomnographic studies. Successful treatment of the OSA with continuous positive airway pressure resulted in permanent stabilization of the hemoglobin to levels below 13 g/dL without the need for repeated phlebotomies and in dramatic lowering of serum erythropoietin levels. To our knowledge, this is the first case of OSA mediated erythrocytosis in a dialysis patient documented in the literature.