Hyland R H, Hutcheon M A, Perl A, Bowes G, Anthonisen N R, Zamel N, Phillipson E A
Am Rev Respir Dis. 1981 Aug;124(2):180-5. doi: 10.1164/arrd.1981.124.2.180.
This report describes a patient with primary alveolar hypoventilation who, after 2 yr of successful treatment with nocturnal oxygen, developed severe hypoxemia and hypercapnia during sleep, morning headaches, and daytime fatigue. Sleep studies demonstrated prolonged periods of hypoventilation and apnea without evidence of upper airway occlusion. Therefore, a phrenic nerve stimulator was implanted to allow pacing of the diaphragm during sleep. However, diaphragm pacing was accompanied by paradoxical movement of the rib cage and upper airway occlusion during sleep, and was unsuccessful in maintaining adequate ventilation. Therefore, the patient underwent a tracheostomy after which diaphragm pacing maintained adequate nocturnal ventilation; however, paradoxical movement of the rib cage persisted. The induction of upper airway occlusion as a result of diaphragm pacing, in contrast to the absence of occlusion during spontaneous breathing, highlights the importance of the normal temporal coordination of inspiratory activation of the upper airway muscles and diaphragm. The findings have important implications for the pathogenesis of obstructive sleep apneas in general.
本报告描述了一名患有原发性肺泡低通气的患者，在夜间吸氧成功治疗2年后，睡眠期间出现严重低氧血症和高碳酸血症、晨起头痛和日间疲劳。睡眠研究显示存在长时间的低通气和呼吸暂停，无气道上段阻塞的证据。因此，植入了膈神经刺激器，以便在睡眠期间对膈肌进行起搏。然而，膈肌起搏伴随着睡眠期间胸廓的反常运动和气道上段阻塞，未能成功维持足够的通气。因此，该患者接受了气管切开术，之后膈肌起搏维持了足够的夜间通气；然而，胸廓的反常运动仍然存在。与自主呼吸时无阻塞相反，膈肌起搏导致气道上段阻塞，这突出了上气道肌肉和膈肌吸气激活正常时间协调的重要性。这些发现对一般阻塞性睡眠呼吸暂停的发病机制具有重要意义。
