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Vagus nerve stimulator (VNS)-associated sleep disordered breathing (SDB) is a poorly understood side effect of VNS treatment. We present a patient with VNS-associated SDB who underwent sleep laboratory VNS titration, evaluation by drug-induced sleep endoscopy, and treatment including adenotonsillectomy and positive airway pressure therapy. This case is unique as it is the first to document, in real time, the inverse correlation of VNS current with airflow. This case offers unique insights into mechanisms and treatment of VNS-associated SDB, and a novel approach in its management. Most importantly, this case highlights the need for collaboration between physicians managing epilepsy with VNS and those managing sleep.