>Rationale: 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.>Objectives: The objective of our study was to evaluate the cost-effectiveness of HRCT chest imaging to facilitate early diagnosis of LAM, BHD, and PLCH.>Methods: 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.>Measurements and Main Results: In our base case analysis, screeningfor the presence of BHD, LAM, or PLCH in patients presenting with a spontaneouspneumothorax was cost effective, with a marginal cost-effectiveness ratio of$1,427 per quality-adjusted life-year gained. Sensitivity analysis showed thatscreening HRCT remained cost effective for diffuse cystic lung diseasesprevalence as low as 0.01%.>Conclusions: HRCT image screening for BHD, LAM, and PLCH in patientswith apparent primary spontaneous pneumothorax is cost effective. Cliniciansshould consider performing a screening HRCT in patients presenting with apparentprimary spontaneous pneumothorax.
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