Abstract

Abstract Introduction Primary central sleep apnea of infancy tends to improve over weeks with supportive care. No established treatments exist; however, infants with this condition remain at risk from sequelae of intermittent hypoxemia. We present a term infant with primary central sleep apnea of infancy treated with caffeine citrate resulting in clinical and polysomnographic improvement. Report of Cases: A 7-day-old male infant born at 37 weeks gestation (gestational age confirmed by early first trimester prenatal ultrasound) was hospitalized following an episode of hypotonia and decreased responsiveness. Infectious studies, chest radiograph, and echocardiogram were normal. During the hospitalization, oxygen desaturations during sleep were observed and capillary blood gas during sleep showed a pH of 7.36 and a partial pressure of carbon dioxide of 54 mmHg. Polysomnography on room air showed central sleep apnea [central apnea hypopnea index (AHI) of 58, obstructive AHI of 4, hypoxemia, hypoventilation with transcutaneous carbon dioxide greater than 50 mmHg for 89% of sleep time, and periodic breathing for 21.7% of sleep time]. Brain MRI and paired-like homeobox2B (PHOX2B) genetic testing were normal. A trial of caffeine citrate was initiated with prompt resolution of oxygen desaturations during sleep. Serial capillary blood gases showed improvement with partial pressure of carbon dioxide between 39-44 mmHg. Polysomnography on room air three days after caffeine initiation demonstrated resolved hypoxemia, hypoventilation, periodic breathing, obstructive sleep apnea, and central sleep apnea (central AHI of 6.8). The patient was discharged home on caffeine and continuous pulse oximetry during sleep. At follow-up six weeks later, the patient had no oxygen desaturations and was successfully weaned off caffeine. Conclusion To our knowledge, there are no prior reports of term infants being treated with caffeine citrate for primary central sleep apnea of infancy. While caffeine is an established therapy for apnea of prematurity, it is typically discontinued at a postmenstrual age of 32 - 34 weeks. Our case demonstrates that in term infants with no underlying medical conditions and primary central sleep apnea of infancy, immature regulation of respiration should be suspected, and a trial of caffeine may be considered. Support (If Any) None.

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