Disease homogeneity and treatment heterogeneity in idiopathic subglottic stenosis.
Disease homogeneity and treatment heterogeneity in idiopathic subglottic stenosis.
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DOI：
10.1002/lary.25708
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发表时间：
2016-06
期刊：
影响因子：
--
通讯作者：
Francis DO
中科院分区：
文献类型：
--
作者：
Gelbard A;Donovan DT;Ongkasuwan J;Nouraei SA;Sandhu G;Benninger MS;Bryson PC;Lorenz RR;Tierney WS;Hillel AT;Gadkaree SK;Lott DG;Edell ES;Ekbom DC;Kasperbauer JL;Maldonado F;Schindler JS;Smith ME;Daniero JJ;Garrett CG;Netterville JL;Rickman OB;Sinard RJ;Wootten CT;Francis DO
特发性声门下狭窄（iSGS）是一种罕见且可能危及生命的疾病，其特征是经常需要重复手术以稳定气道的复发性和进行性气道阻塞。病因不明和低患病率限制了描述iSGS自然史的能力，并导致手术管理的可变性。尚不确定这种变化与临床结局的关系。病历摘要。利用国际多机构合作，我们收集了有关患者特征、治疗和临床结局的回顾性数据。我们研究了开放式和内窥镜治疗方法之间的差异，并评估了治疗结果;特别是，疾病复发和末次随访时是否需要气管造口术。值得注意的是，10个参与中心的479例iSGS患者几乎全部为女性（98%，95%置信区间[CI]，96.1-99.6）、高加索人（95%，95% CI，92.2-98.8）和其他健康人群（平均年龄校正的Charlson合并症指数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%的患者接受了内镜治疗，而19.8%的患者接受了开放性重建。内镜手术的疾病复发率显著高于开放手术（卡方= 4.09，P = 0.043）。无论手术入路如何，97%的患者避免了气管切开术（95% CI，94.5-99.8）。有趣的是，使用相似治疗方法的中心之间的疾病复发率存在异常值。特发性声门下狭窄患者的同质性令人惊讶。治疗方法的异质性和观察到的中心之间疾病复发率的离群值提高了通过详细了解护理过程改善临床结局的可能性。
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. Medical record abstraction. 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. 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 (chi2 = 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. 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.
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