Inglis A F, Kokesh J, Siebert J, Richardson M A
Department of Otolaryngology, Head and Neck Surgery, University of Washington, Seattle.
Arch Otolaryngol Head Neck Surg. 1992 Apr;118(4):436-8. doi: 10.1001/archotol.1992.01880040102017.
Anterior vertical fusion of the tracheal cartilages with normal pars membranacea is a rarely described tracheal anomaly. We report four cases of this anomaly, three associated with craniosynostosis syndromes and one with Goldenhar's syndrome. The anomaly was documented at postmortem examination in one case, both endoscopically and at tracheotomy in two cases, and endoscopically in one case. Documentation from gross postmortem photographs, photomicrographs, and intraoperative endoscopic photographs is presented so that the anatomical aspects of this anomaly can be thoroughly understood and recognized. All four newborn patients developed recurrent lower respiratory tract infections, reactive airway disease, and chronically retained secretions attributable to this anomaly. Neither tracheal stenosis nor stridor was encountered. In the three surviving patients, the infections grew less frequent and less severe in later childhood. These observations suggest that this tracheobronchial anomaly may be associated with a variety of craniofacial syndromes and may alter airflow dynamics in a way that predisposes to retained secretions and chronic pulmonary disease.
气管软骨与正常膜部的前垂直融合是一种鲜有描述的气管异常。我们报告了4例这种异常病例,其中3例与颅缝早闭综合征相关,1例与Goldenhar综合征相关。该异常在1例尸检时被记录,2例通过内镜检查和气管切开术记录,1例通过内镜检查记录。文中展示了大体尸检照片、显微照片和术中内镜照片,以便能全面理解和认识这种异常的解剖学特征。所有4例新生儿患者均因这种异常出现反复下呼吸道感染、反应性气道疾病和长期分泌物潴留。未发现气管狭窄或喘鸣。在3例存活患者中,儿童后期感染的频率和严重程度均有所降低。这些观察结果表明,这种气管支气管异常可能与多种颅面综合征相关,并可能以一种易导致分泌物潴留和慢性肺部疾病的方式改变气流动力学。