Health Research Center, Baqiyatallah University of Medical Sciences, Tehran, Iran.
Paediatr Respir Rev. 2013 Dec;14(4):250-5. doi: 10.1016/j.prrv.2013.02.009. Epub 2013 Mar 21.
Respiratory difficulties and breathing disorders in achondroplasia are thought to underlie the increased risk for sudden infant death and neuropsychological deficits seen in this condition. This review evaluates literature regarding respiratory dysfunctions and their sequelae in patients with achondroplasia. The limited number of prospective studies of respiratory disease in achondroplasia means that observational studies and case series provide a large proportion of the data regarding the spectrum of respiratory diseases in achondroplasia and their treatments. Amongst clinical respiratory problems described, snoring is the commonest observed abnormality, but the reported incidence of obstructive sleep apnoea (OSA) shows wide variance (10% to 75%). Reported treatments of OSA include adenotonsillectomy, the use of CPAP, and surgical improvement of the airway, including mid-face advancement. Otolaryngologic manifestations are also common. Respiratory failure due to small thoracic volumes is reported, but uncommon. Mortality rate at all ages was 2.27 (CI: 1.7-3.0) with age-specific mortality increased at all ages. Sudden death was most common in infants and children. Cardiovascular events are the main cause of mortality in adults. Despite earlier recognition and treatment of respiratory complications of achondroplasia, increased mortality rates and other complications remain high. Future and ongoing evaluation of the prevalence and impact of respiratory disorders, particularly OSA, in achondroplasia is recommended.
人们认为,软骨发育不全症患者存在呼吸困难和呼吸障碍,这是导致这种情况下婴儿猝死和神经心理缺陷风险增加的原因。本综述评估了软骨发育不全症患者呼吸功能障碍及其后果的相关文献。由于软骨发育不全症患者的呼吸系统疾病的前瞻性研究数量有限,因此观察性研究和病例系列研究为软骨发育不全症患者的呼吸系统疾病谱及其治疗方法提供了大量数据。在描述的临床呼吸问题中,打鼾是最常见的异常,但阻塞性睡眠呼吸暂停(OSA)的报告发病率差异很大(10%至 75%)。报道的 OSA 治疗方法包括腺样体扁桃体切除术、使用 CPAP 以及包括中面部提升术在内的气道手术改善。耳鼻喉科表现也很常见。由于胸腔容积小而导致呼吸衰竭的情况也有报道,但并不常见。所有年龄段的死亡率为 2.27(CI:1.7-3.0),所有年龄段的特定年龄死亡率都有所增加。婴儿和儿童最常见的是突然死亡。心血管事件是成年人死亡的主要原因。尽管对软骨发育不全症的呼吸并发症的早期认识和治疗有所增加,但死亡率和其他并发症仍然很高。建议未来对软骨发育不全症的呼吸障碍,特别是 OSA 的流行程度和影响进行评估。