{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,30]],"date-time":"2026-05-30T02:43:35Z","timestamp":1780109015212,"version":"3.54.0"},"reference-count":23,"publisher":"MDPI AG","issue":"14","license":[{"start":{"date-parts":[[2024,7,15]],"date-time":"2024-07-15T00:00:00Z","timestamp":1721001600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Division of Intramural Research, National Heart, Lung and Blood Institute, NIH Internal Research Program, the National Institutes of Health, USA","award":["HL006141"],"award-info":[{"award-number":["HL006141"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Diagnostics"],"abstract":"<jats:p>In cystic lung diseases such as lymphangioleiomyomatosis (LAM), a CT-based cyst score that measures the percentage of the lung volume occupied by cysts is a common index of the cyst burden in the lungs. Although the current semi-automatic measurement of the cyst score is well established, it is susceptible to human operator variabilities. We recently developed a fully automatic method incorporating adaptive features in place of manual adjustments. In this clinical study, the automatic method is validated against the standard method in several aspects. These include the agreement between the cyst scores of the two methods, the agreement of each method with independent tests of pulmonary function, and the temporal consistency of the measurements in the consecutive visits of the same patients. We found that the automatic method agreed with the standard method as well as the agreement between two trained operators running the same standard method; both methods obtained the same level of correlation with laboratory pulmonary function tests; the automated method had better temporal consistency than the standard method (p &lt; 0.0001). The study indicates that the automatic method could replace the standard method and provide better consistency in assessing the extent of cystic changes in the lungs of patients.<\/jats:p>","DOI":"10.3390\/diagnostics14141529","type":"journal-article","created":{"date-parts":[[2024,7,15]],"date-time":"2024-07-15T18:17:32Z","timestamp":1721067452000},"page":"1529","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Clinical Trial Validation of Automated Segmentation and Scoring of Pulmonary Cysts in Thoracic CT Scans"],"prefix":"10.3390","volume":"14","author":[{"given":"Aneesha","family":"Baral","sequence":"first","affiliation":[{"name":"Laboratory of Imaging Physics, Biochemistry and Biophysics Center, National Heart, Lung and Blood Institute, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Simone","family":"Lee","sequence":"additional","affiliation":[{"name":"Laboratory of Imaging Physics, Biochemistry and Biophysics Center, National Heart, Lung and Blood Institute, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Farah","family":"Hussaini","sequence":"additional","affiliation":[{"name":"Laboratory of Imaging Physics, Biochemistry and Biophysics Center, National Heart, Lung and Blood Institute, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brianna","family":"Matthew","sequence":"additional","affiliation":[{"name":"Laboratory of Imaging Physics, Biochemistry and Biophysics Center, National Heart, Lung and Blood Institute, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9581-5776","authenticated-orcid":false,"given":"Alfredo","family":"Lebron","sequence":"additional","affiliation":[{"name":"Laboratory of Imaging Physics, Biochemistry and Biophysics Center, National Heart, Lung and Blood Institute, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Muyang","family":"Wang","sequence":"additional","affiliation":[{"name":"Laboratory of Imaging Physics, Biochemistry and Biophysics Center, National Heart, Lung and Blood Institute, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0826-7290","authenticated-orcid":false,"given":"Li-Yueh","family":"Hsu","sequence":"additional","affiliation":[{"name":"Department of Radiology and Imaging Sciences, Clinical Center, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joel","family":"Moss","sequence":"additional","affiliation":[{"name":"Pulmonary Branch, National Heart, Lung and Blood Institute, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6844-2997","authenticated-orcid":false,"given":"Han","family":"Wen","sequence":"additional","affiliation":[{"name":"Laboratory of Imaging Physics, Biochemistry and Biophysics Center, National Heart, Lung and Blood Institute, National Institutes of Health, Bethesda, MD 20892, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2024,7,15]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"971","DOI":"10.1093\/qjmed\/hcr116","article-title":"Use of Variability in National and Regional Data to Estimate the Prevalence of Lymphangioleiomyomatosis","volume":"104","author":"Harknett","year":"2011","journal-title":"QJM"},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1313","DOI":"10.1016\/S2213-2600(21)00228-9","article-title":"Lymphangioleiomyomatosis: Pathogenesis, Clinical Features, Diagnosis, and Management","volume":"9","author":"McCarthy","year":"2021","journal-title":"Lancet Respir. Med."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"319","DOI":"10.1016\/j.ejim.2007.10.015","article-title":"Lymphangioleiomyomatosis: A Review","volume":"19","author":"Hohman","year":"2008","journal-title":"Eur. J. Intern. Med."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"20","DOI":"10.1007\/s10038-002-8651-8","article-title":"Mutation Analysis of the TSC1 and TSC2 Genes in Japanese Patients with Pulmonary Lymphangioleiomyomatosis","volume":"47","author":"Sato","year":"2002","journal-title":"J. Hum. Genet."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"1354","DOI":"10.1164\/rccm.201411-2094CI","article-title":"Diffuse Cystic Lung Disease. Part I","volume":"191","author":"Gupta","year":"2015","journal-title":"Am. J. Respir. Crit. Care Med."},{"key":"ref_6","unstructured":"Khaddour, K., Sankari, A., and Shayuk, M. (2024). Lymphangioleiomyomatosis. StatPearls, StatPearls Publishing."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"875","DOI":"10.1136\/thx.2004.035154","article-title":"Emerging Clinical Picture of Lymphangioleiomyomatosis","volume":"60","author":"Cohen","year":"2005","journal-title":"Thorax"},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1007\/s00408-012-9419-3","article-title":"Clinical Predictors of Mortality and Cause of Death in Lymphangioleiomyomatosis: A Population-Based Registry","volume":"191","author":"Oprescu","year":"2013","journal-title":"Lung"},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"14","DOI":"10.1183\/09031936.00076209","article-title":"European Respiratory Society Guidelines for the Diagnosis and Management of Lymphangioleiomyomatosis","volume":"35","author":"Johnson","year":"2010","journal-title":"Eur. Respir. J."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"748","DOI":"10.1164\/rccm.201607-1384ST","article-title":"Official American Thoracic Society\/Japanese Respiratory Society Clinical Practice Guidelines: Lymphangioleiomyomatosis Diagnosis and Management","volume":"194","author":"McCormack","year":"2016","journal-title":"Am. J. Respir. Crit. Care Med."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"381","DOI":"10.1148\/radiology.176.2.2367651","article-title":"Lymphangiomyomatosis\u2014CT, Chest Radiographic, and Functional Correlations","volume":"176","author":"Aberle","year":"1990","journal-title":"Radiology"},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"342","DOI":"10.1513\/AnnalsATS.201509-631OC","article-title":"A Novel Quantitative Computed Tomographic Analysis Suggests How Sirolimus Stabilizes Progressive Air Trapping in Lymphangioleiomyomatosis","volume":"13","author":"Argula","year":"2016","journal-title":"Ann. Am. Thorac. Soc."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"199","DOI":"10.1016\/j.chest.2021.01.077","article-title":"Ultra-Small Lung Cysts Impair Diffusion Without Obstructing Air Flow in Lymphangioleiomyomatosis","volume":"160","author":"Matthew","year":"2021","journal-title":"Chest"},{"key":"ref_14","doi-asserted-by":"crossref","unstructured":"Lee, S., Lebron, A., Matthew, B., Moss, J., and Wen, H. (2023). Automated Segmentation and Measurements of Pulmonary Cysts in Lymphangioleiomyomatosis across Multiple CT Scanner Platforms over a Period of Two Decades. Bioengineering, 10.","DOI":"10.3390\/bioengineering10111255"},{"key":"ref_15","doi-asserted-by":"crossref","first-page":"1037","DOI":"10.2214\/AJR.07.3334","article-title":"Automated Algorithm for Quantifying the Extent of Cystic Change on Volumetric Chest CT: Initial Results in Lymphangioleiomyomatosis","volume":"192","author":"Schmithorst","year":"2009","journal-title":"Am. J. Roentgenol."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"1324","DOI":"10.1183\/09031936.00080312","article-title":"Multi-Ethnic Reference Values for Spirometry for the 3\u201395-Yr Age Range: The Global Lung Function 2012 Equations","volume":"40","author":"Quanjer","year":"2012","journal-title":"Eur. Respir. J."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"1700010","DOI":"10.1183\/13993003.00010-2017","article-title":"Official ERS Technical Standards: Global Lung Function Initiative Reference Values for the Carbon Monoxide Transfer Factor for Caucasians","volume":"50","author":"Stanojevic","year":"2017","journal-title":"Eur. Respir. J."},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"353","DOI":"10.1148\/radiology.144.2.7089289","article-title":"Two Methods for Isolating the Lung Area of a CT Scan for Density Information","volume":"144","author":"Hedlund","year":"1982","journal-title":"Radiology"},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"574","DOI":"10.1164\/ajrccm.161.2.9812073","article-title":"An Analysis Algorithm for Measuring Airway Lumen and Wall Areas from High-Resolution Computed Tomographic Data","volume":"161","author":"King","year":"2000","journal-title":"Am. J. Respir. Crit. Care Med."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"307","DOI":"10.1016\/S0140-6736(86)90837-8","article-title":"Statistical-Methods for Assessing Agreement between 2 Methods of Clinical Measurement","volume":"1","author":"Bland","year":"1986","journal-title":"Lancet"},{"key":"ref_21","unstructured":"Lee, I.A., and Preacher, K.J. (2024, June 06). Calculation for the Test of the Difference between Two Dependent Correlations with One Variable in Common [Computer Software]. Available online: https:\/\/quantpsy.org\/corrtest\/corrtest2.htm."},{"key":"ref_22","doi-asserted-by":"crossref","unstructured":"Kufel, J., Biel\u00f3wka, M., Rojek, M., Mitr\u0119ga, A., Lewandowski, P., Cebula, M., Krawczyk, D., Biel\u00f3wka, M., Kondo\u0142, D., and Bargie\u0142-\u0141\u0105czek, K. (2023). Multi-Label Classification of Chest X-Ray Abnormalities Using Transfer Learning Techniques. J. Pers. Med., 13.","DOI":"10.3390\/jpm13101426"},{"key":"ref_23","doi-asserted-by":"crossref","unstructured":"Zhang, L., Gopalakrishnan, V., Lu, L., Summers, R.M., Moss, J., and Yao, J. (2018). Self-Learning to Detect and Segment Cysts in Lung CT Images without Manual Annotation. arXiv.","DOI":"10.1109\/ISBI.2018.8363763"}],"container-title":["Diagnostics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2075-4418\/14\/14\/1529\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T15:17:22Z","timestamp":1760109442000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2075-4418\/14\/14\/1529"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,7,15]]},"references-count":23,"journal-issue":{"issue":"14","published-online":{"date-parts":[[2024,7]]}},"alternative-id":["diagnostics14141529"],"URL":"https:\/\/doi.org\/10.3390\/diagnostics14141529","relation":{},"ISSN":["2075-4418"],"issn-type":[{"value":"2075-4418","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,7,15]]}}}