Matsuyama Masashi, Nakazawa Kensuke, Katou Minoru, Ota Kyoko, Masuko Hironori, Iizuka Takashi, Mori Takeru, Hayashi Hiroki, Hayashihara Kenji, Saito Takefumi, Satoh Makoto, Hizawa Nobuyuki
Department of Respiratory Medicine, National Hospital Organization Ibarakihigashi National Hospital, Ibaraki.
Intern Med. 2009;48(11):925-30. doi: 10.2169/internalmedicine.48.1804. Epub 2009 Jun 1.
A 29-year-old man with a history of resected bulbar hemangioblastoma was admitted to hospital with nighttime breathing disturbance, but with apparently normal breathing while awake. After diagnostic work-up, including polysomnographic testing, he was diagnosed as having central alveolar hypoventilation syndrome due to surgical resection for bulbar hemangioblastoma. Non-invasive positive pressure ventilation (NIPPV) via oronasal facemask was given for nocturnal ventilatory support. Two months after leaving our hospital, he was readmitted because of aspiration pneumonia. The pneumonia was successfully treated with antibiotics, but the desaturation during sleep worsened despite non-invasive ventilatory support. Higher bi-level positive pressure using a full facemask successfully alleviated sleep hypoventilation and apnea. To the best of our knowledge, this is the first case report of central alveolar hypoventilation syndrome due to surgical resection for bulbar hemangioblastoma.
一名有延髓血管母细胞瘤切除病史的29岁男性因夜间呼吸障碍入院,但清醒时呼吸明显正常。经过包括多导睡眠图测试在内的诊断检查后,他被诊断为因延髓血管母细胞瘤手术切除导致的中枢性肺泡低通气综合征。通过口鼻面罩给予无创正压通气(NIPPV)进行夜间通气支持。出院两个月后,他因吸入性肺炎再次入院。肺炎通过抗生素成功治疗,但尽管有无创通气支持,睡眠期间的低氧血症仍恶化。使用全面罩的更高双水平正压成功缓解了睡眠低通气和呼吸暂停。据我们所知,这是首例因延髓血管母细胞瘤手术切除导致中枢性肺泡低通气综合征的病例报告。