1 Division of Pulmonary, Critical Care, and Sleep Medicine, Department of Internal Medicine.
2 Department of Environmental Heath, and.
Ann Am Thorac Soc. 2017 Jan;14(1):17-25. doi: 10.1513/AnnalsATS.201606-459OC.
Patients without a known history of lung disease presenting with a spontaneous pneumothorax are generally diagnosed as having primary spontaneous pneumothorax. However, occult diffuse cystic lung diseases such as Birt-Hogg-Dubé syndrome (BHD), lymphangioleiomyomatosis (LAM), and pulmonary Langerhans cell histiocytosis (PLCH) can also first present with a spontaneous pneumothorax, and their early identification by high-resolution computed tomographic (HRCT) chest imaging has implications for subsequent management.
The objective of our study was to evaluate the cost-effectiveness of HRCT chest imaging to facilitate early diagnosis of LAM, BHD, and PLCH.
We constructed a Markov state-transition model to assess the cost-effectiveness of screening HRCT to facilitate early diagnosis of diffuse cystic lung diseases in patients presenting with an apparent primary spontaneous pneumothorax. Baseline data for prevalence of BHD, LAM, and PLCH and rates of recurrent pneumothoraces in each of these diseases were derived from the literature. Costs were extracted from 2014 Medicare data. We compared a strategy of HRCT screening followed by pleurodesis in patients with LAM, BHD, or PLCH versus conventional management with no HRCT screening.
In our base case analysis, screening for the presence of BHD, LAM, or PLCH in patients presenting with a spontaneous pneumothorax was cost effective, with a marginal cost-effectiveness ratio of $1,427 per quality-adjusted life-year gained. Sensitivity analysis showed that screening HRCT remained cost effective for diffuse cystic lung diseases prevalence as low as 0.01%.
HRCT image screening for BHD, LAM, and PLCH in patients with apparent primary spontaneous pneumothorax is cost effective. Clinicians should consider performing a screening HRCT in patients presenting with apparent primary spontaneous pneumothorax.
无肺部疾病既往史的自发性气胸患者通常被诊断为原发性自发性气胸。然而,隐匿性弥漫性肺囊性疾病,如 Birt-Hogg-Dubé 综合征(BHD)、淋巴管平滑肌瘤病(LAM)和肺朗格汉斯细胞组织细胞增生症(PLCH),也可能首先表现为自发性气胸,通过高分辨率计算机断层扫描(HRCT)胸部成像早期识别这些疾病对后续治疗具有重要意义。
本研究旨在评估 HRCT 胸部成像的成本效益,以促进 LAM、BHD 和 PLCH 的早期诊断。
我们构建了一个马尔可夫状态转移模型,以评估 HRCT 筛查在促进表现为明显原发性自发性气胸的患者中早期诊断弥漫性囊性肺疾病的成本效益。BHD、LAM 和 PLCH 的患病率以及这些疾病中复发性气胸的发生率的基线数据来自文献。成本从 2014 年医疗保险数据中提取。我们比较了在 LAM、BHD 或 PLCH 患者中进行 HRCT 筛查后行胸膜固定术与不进行 HRCT 筛查的常规治疗策略。
在我们的基本情况分析中,对自发性气胸患者进行 BHD、LAM 或 PLCH 筛查具有成本效益,每获得一个质量调整生命年的边际成本效益比为 1427 美元。敏感性分析表明,对于弥漫性囊性肺疾病的患病率低至 0.01%,HRCT 筛查仍然具有成本效益。
在表现为明显原发性自发性气胸的患者中,对 BHD、LAM 和 PLCH 进行 HRCT 图像筛查具有成本效益。临床医生应考虑对表现为明显原发性自发性气胸的患者进行筛查性 HRCT。