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Abstract Introduction Arterial tortuosity has emerged as a predictor of adverse outcomes in congenital aortopathies using 3D reconstructed images. We validated a new method to estimate aortic arch tortuosity on 2D CT. We hypothesize that arch tortuosity may identify bicuspid aortic valve (BAV) patients at high risk to develop thoracic aortic aneurysms or aortic dissections (TAD). Methods BAV subjects with chest CT scans were retrospectively identified in our clinical records and matched to tricuspid aortic valve (TAV) controls by age, gender, and presentation with TAD. Subjects with prior ascending aortic intervention were excluded. Measurements included aortic arch tortuosity, length, angle, width and height. Total aortic tortuosity was estimated in subjects with available abdominal images. Results 120 BAV and 234 TAV subjects were included. Our 2D measurements were highly correlated with 3D midline arch measurements and had high inter- and intra-observer reliability. Compared to TAV, BAV subjects had increased arch tortuosity (median 1.76 [Q1-Q3: 1.62–1.95] vs. 1.63 [1.53–1.78], P < 0.01), length (149 [136–160] vs. 135 [122–152] mm, P < 0.01), height (46 [41–53] vs. 39 [34–47] mm, P < 0.01), and vertex acuity (70 [61–77] vs. 75 [68–81] degree, P < 0.01). In a multivariable analysis, arch tortuosity remained independently associated with BAV after adjusting for aortic diameter and other clinical characteristics. Conclusions We found that aortic arch tortuosity is significantly increased in BAV and may identify BAV patients who are at increased risk for TAD. Further studies to evaluate the association between tortuosity and clinical outcomes are in progress. Highlights • We validated a new method to measure aortic arch tortuosity, a biomarker of risk for thoracic aortic disease • Our new method will greatly expand the number of images that are accessible to tortuosity measurements • Arch tortuosity may identify a high-risk subgroup of bicuspid aortic valve patients and is independent of other risk factors [ABSTRACT FROM AUTHOR]