Stater Brian J, Oomen Karin P Q, Modi Vikash K
Division of Pediatric Otolaryngology, Department of Otolaryngology-Head and Neck Surgery, Weill Cornell Medical College, New York, New York.
Division of Pediatric Otolaryngology, Department of Otolaryngology-Head and Neck Surgery, University Medical Center Utrecht-Wilhelmina Children's Hospital, Utrecht, the Netherlands.
JAMA Otolaryngol Head Neck Surg. 2015 Jan;141(1):73-7. doi: 10.1001/jamaoto.2014.2790.
We report 2 new cases of tracheal cartilaginous sleeve and perform a systematic literature review of all documented cases of this condition. We aim to increase awareness of this anomaly and provide recommendations for both intraoperative and postoperative management of patients with tracheal cartilaginous sleeve undergoing tracheostomy.
We studied the clinical records of 2 children with tracheal cartilaginous sleeves and short tracheas treated at our institution. One of these patients had Beare-Stevenson syndrome, and the other had Crouzon syndrome. Both patients required tracheostomy for persistent upper airway obstruction, and both required custom-length tracheostomy tubes. Bronchoscopy and needle localization were beneficial in performing tracheostomy on these patients. All documented cases of tracheal cartilaginous sleeve in the literature were found to be associated with midface hypoplasia secondary to syndromic craniosynostosis. Seventy-five percent of cases required tracheostomy.
Tracheal cartilaginous sleeve can be associated with Beare-Stevenson syndrome. In infants with midface hypoplasia associated with a craniosynostosis syndrome undergoing tracheostomy, the surgeon must be prepared to encounter and manage tracheal cartilaginous sleeve intraoperatively. Bronchoscopy and needle localization can be beneficial when performing tracheostomy in these patients. Customized, shorter-length tracheostomy tubes should be considered for these patients.
我们报告了2例气管软骨套的新病例，并对所有已记录的该病症病例进行了系统的文献综述。我们旨在提高对这种异常情况的认识，并为接受气管切开术的气管软骨套患者提供术中及术后管理的建议。
我们研究了在我们机构接受治疗的2例患有气管软骨套和短气管的儿童的临床记录。其中1例患者患有贝-史蒂文森综合征，另1例患有克鲁宗综合征。两名患者均因持续性上气道梗阻需要进行气管切开术，且均需要定制长度的气管切开套管。支气管镜检查和针定位有助于对这些患者进行气管切开术。文献中所有已记录的气管软骨套病例均与综合征性颅缝早闭继发的面中部发育不全有关。75%的病例需要进行气管切开术。
气管软骨套可能与贝-史蒂文森综合征有关。对于因颅缝早闭综合征而面中部发育不全的婴儿在进行气管切开术时，外科医生必须做好术中遇到并处理气管软骨套的准备。在这些患者进行气管切开术时，支气管镜检查和针定位可能会有帮助。对于这些患者，应考虑使用定制的较短长度的气管切开套管。
