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埃勒斯-当洛综合征的呼吸系统表现。

Respiratory manifestations in the Ehlers-Danlos syndromes.

作者信息

Bascom Rebecca, Dhingra Radha, Francomano Clair A

机构信息

Division of Pulmonary, Allergy, and Critical Care, Department of Medicine, Penn State College of Medicine, Milton S. Hershey Medical Center, Hershey, Pennsylvania, USA.

Division of Epidemiology, Department of Public Health Sciences, Penn State College of Medicine, Hershey, Pennsylvania, USA.

出版信息

Am J Med Genet C Semin Med Genet. 2021 Dec;187(4):533-548. doi: 10.1002/ajmg.c.31953. Epub 2021 Nov 22.

Abstract

Persons with the Ehlers-Danlos syndromes (EDS) report a wide range of respiratory symptoms, most commonly shortness of breath, exercise limitation, and cough. Also reported are noisy breathing attributed to asthma, difficulty with deep inhalation, and inspiratory thoracic pain. The literature consists of case reports and small cross-sectional and cohort studies. One case-control study estimated twofold to threefold greater respiratory disease burden among persons with EDS as compared to controls. The differential diagnosis for symptoms is broad. Structural alterations include pectus deformities, scoliosis, recurrent rib subluxations, and tracheobronchomalacia, associated with varying degrees of physiologic impairment. Those with vascular EDS have an increased risk of pneumothorax, intrapulmonary bleeding, cysts, and nonmalignant fibrous nodules. Functional aerodigestive manifestations such as inducible laryngeal obstruction may be misdiagnosed as asthma, with gastro-esophageal dysmotility and reflux as common contributing factors. Inflammatory manifestations include costochondritis, bronchiectasis, and localized respiratory allergic and nonallergic mast cell activation. Cranio-cervical instability can dysregulate respiratory control pathways. There is a need for careful phenotyping using standardized clinical tools and patient-reported outcomes and continuing collaboration with aerodigestive specialists including otolaryngologists and gastroenterologists. Also needed is further evaluation of respiratory symptoms in persons with hypermobility spectrum disorders. Personalized monitoring strategies are invaluable for interpretation and long-term management of respiratory symptoms.

摘要

患有埃勒斯-当洛综合征(EDS)的人报告了广泛的呼吸道症状,最常见的是呼吸急促、运动受限和咳嗽。还报告了归因于哮喘的呼吸声、深吸气困难和吸气时胸痛。文献包括病例报告以及小型横断面研究和队列研究。一项病例对照研究估计,与对照组相比,EDS患者的呼吸道疾病负担高出两到三倍。症状的鉴别诊断范围很广。结构改变包括漏斗胸畸形、脊柱侧弯、复发性肋骨半脱位和气管支气管软化,并伴有不同程度的生理功能损害。患有血管型EDS的人发生气胸、肺内出血、囊肿和非恶性纤维结节的风险增加。功能性气消化道表现,如可诱导性喉梗阻,可能被误诊为哮喘,胃食管动力障碍和反流是常见的促成因素。炎症表现包括肋软骨炎、支气管扩张以及局部呼吸道过敏和非过敏性肥大细胞活化。颅颈不稳定可导致呼吸控制途径失调。需要使用标准化临床工具和患者报告的结果进行仔细的表型分析,并继续与包括耳鼻喉科医生和胃肠病学家在内的气消化道专家合作。还需要对高活动度谱系障碍患者的呼吸道症状进行进一步评估。个性化监测策略对于呼吸道症状的解释和长期管理非常重要。

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