特发性声门下狭窄的疾病同质性与治疗异质性
Disease homogeneity and treatment heterogeneity in idiopathic subglottic stenosis.
作者信息
Gelbard Alexander, Donovan Donald T, Ongkasuwan Julina, Nouraei S A R, Sandhu Guri, Benninger Michael S, Bryson Paul C, Lorenz Robert R, Tierney William S, Hillel Alexander T, Gadkaree Shekhar K, Lott David G, Edell Eric S, Ekbom Dale C, Kasperbauer Jan L, Maldonado Fabien, Schindler Joshua S, Smith Marshall E, Daniero James J, Garrett C Gaelyn, Netterville James L, Rickman Otis B, Sinard Robert J, Wootten Christopher T, Francis David O
机构信息
Vanderbilt University Medical Center Dept. of Otolaryngology and Pulmonology, Nashville, Tennessee.
Baylor College of Medicine Dept. of Otolaryngology, Houston, Texas.
出版信息
Laryngoscope. 2016 Jun;126(6):1390-6. doi: 10.1002/lary.25708. Epub 2015 Nov 4.
OBJECTIVES/HYPOTHESIS: Idiopathic subglottic stenosis (iSGS) is a rare and potentially life-threatening disease marked by recurrent and progressive airway obstruction frequently requiring repeated surgery to stabilize the airway. Unknown etiology and low disease prevalence have limited the ability to characterize the natural history of iSGS and resulted in variability in surgical management. It is uncertain how this variation relates to clinical outcomes.
STUDY DESIGN
Medical record abstraction.
METHODS
Utilizing an international, multi-institutional collaborative, we collected retrospective data on patient characteristics, treatment, and clinical outcomes. We investigated variation between and within open and endoscopic treatment approaches and assessed therapeutic outcomes; specifically, disease recurrence and need for tracheostomy at last follow-up.
RESULTS
Strikingly, 479 iSGS patients across 10 participating centers were nearly exclusively female (98%, 95% confidence interval [CI], 96.1-99.6), Caucasian (95%, 95% CI, 92.2-98.8), and otherwise healthy (mean age-adjusted Charlson Comorbidity Index 1.5; 95% CI, 1.44-1.69). The patients presented at a mean age of 50 years (95% CI, 48.8-51.1). A total of 80.2% were managed endoscopically, whereas 19.8% underwent open reconstruction. Endoscopic surgery had a significantly higher rate of disease recurrence than the open approach (chi(2) = 4.09, P = 0.043). Tracheostomy was avoided in 97% of patients irrespective of surgical approach (95% CI, 94.5-99.8). Interestingly, there were outliers in rates of disease recurrence between centers using similar treatment approaches.
CONCLUSION
Idiopathic subglottic stenosis patients are surprisingly homogeneous. The heterogeneity of treatment approaches and the observed outliers in disease recurrence rates between centers raises the potential for improved clinical outcomes through a detailed understanding of the processes of care.
LEVEL OF EVIDENCE
- Laryngoscope, 126:1390-1396, 2016.
目的/假设：特发性声门下狭窄（iSGS）是一种罕见且可能危及生命的疾病，其特征为反复性和进行性气道阻塞，常需反复手术以稳定气道。病因不明和疾病患病率低限制了对iSGS自然病史的描述能力，并导致手术管理的差异。目前尚不确定这种差异与临床结果有何关联。
研究设计
病历摘要。
方法
利用国际多机构合作，我们收集了关于患者特征、治疗和临床结果的回顾性数据。我们调查了开放治疗和内镜治疗方法之间以及内部的差异，并评估了治疗结果；具体而言，是疾病复发情况以及最后一次随访时气管造口术的需求。
结果
引人注目的是，来自10个参与中心的479例iSGS患者几乎全为女性（98%，95%置信区间[CI]，96.1 - 99.6）、白种人（95%，95% CI，92.2 - 98.8），且其他方面健康（平均年龄调整后的查尔森合并症指数为1.5；95% CI，1.44 - 1.69）。患者的平均年龄为50岁（95% CI，48.8 - 51.1）。共有80.2%的患者接受内镜治疗，而19.8%的患者接受开放重建手术。内镜手术的疾病复发率明显高于开放手术（χ² = 4.09，P = 0.043）。无论采用何种手术方式，97%的患者避免了气管造口术（95% CI，94.5 - 99.8）。有趣的是，采用相似治疗方法的中心之间疾病复发率存在异常值。
结论
特发性声门下狭窄患者惊人地同质化。治疗方法的异质性以及各中心疾病复发率中观察到的异常值表明，通过详细了解护理过程，有可能改善临床结果。
证据级别
4。《喉镜》，126:1390 - 1396，2016年。
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