PDF(Pubmed)
Abstract：
Anti-IgLON5 disease is a recently described entity that has been associated with neurological symptoms and sleep disturbances including sleep breathing disorders. Sleep stridor as well as obstructive and less often central sleep apnea have been reported but rarely needing ventilation on tracheotomy. We report the case of a patient in whom obstructive sleep apnea with secondary development of dysphagia and recurrent aspiration pneumonia led to the diagnosis of anti-IgLON 5 disease. Acute respiratory failure due to laryngospasm required intubation and eventually tracheotomy. Yet hypoventilation persisted, and polysomnography demonstrated central sleep apnea alternating with sleep-related tachypnea. Nocturnal ventilation was thus reintroduced. The association of obstructive sleep apnea with dysphagia is a potential red flag for anti-IgLON5 disease, which remains an overlooked diagnosis. Breathing disorders can be complex in this context, with a mixed obstructive and central pattern whose central component can be unveiled after tracheotomy. This highlights the importance of closely monitoring sleep and respiration even after tracheotomy.
Tankéré P, Le Cam P, Folliet L, et al. Unveiled central hypoventilation after tracheotomy in anti-IgLON5 disease: a case report. J Clin Sleep Med. 2023;19(9):1701-1704.
Tankéré P, Le Cam P, Folliet L, et al. Unveiled central hypoventilation after tracheotomy in anti-IgLON5 disease: a case report. J Clin Sleep Med. 2023;19(9):1701-1704.
摘要：
抗IgLON5疾病是最近描述的与神经症状和睡眠障碍(包括睡眠呼吸障碍)相关的实体。据报道，睡眠喘鸣以及阻塞性睡眠呼吸暂停，以及不太常见的中枢睡眠呼吸暂停。但很少需要气管切开术的通气.我们报告了一例阻塞性睡眠呼吸暂停（OSA）伴有吞咽困难和复发性吸入性肺炎的继发性发展导致诊断为抗IgLON5疾病的患者。喉痉挛引起的急性呼吸衰竭需要插管并最终进行气管切开术。然而，通气不足持续存在，多导睡眠图显示中枢睡眠呼吸暂停与睡眠相关的呼吸急促交替。因此重新引入了夜间通气。OSA与吞咽困难的关联是抗Iglon5疾病的潜在危险信号，这仍然是一个被忽视的诊断。在这种情况下，呼吸障碍可能很复杂，具有混合的阻塞性和中央模式，其中央成分可以在气管切开术后显现。这突出了即使在气管切开术后也要密切监测睡眠和呼吸的重要性。
