We report the case of a patient with chronic renal failure and primary hyperparathyroidism who developed nonhypercapnic central sleep apnea syndrome (CSAS), which was multifactorial in origin and attributed to metabolic factors. Given an inadequate response to oxygen therapy and continuous positive airway pressure (CPAP) revealed by several polygraph studies, the patient was treated with bilevel positive airway pressure ventilatory support. Three months after treatment commenced, a parathyroidectomy was performed and hemodialysis was initiated. At this point it was observed that the patient no longer experienced somnolence; moreover, polysomnography revealed partial improvement in the CSAS and normalization of ventilatory patterns on application of nasal CPAP at 7 cm H2O. We discuss the pathogenesis of CSAS associated with chronic kidney failure along with the treatment options and conclude that treatment should be customized due to the lack of predictability of patient response.