Validating a Curvature-Based Marker of Cervical Carotid Tortuosity for Risk Assessment in Heritable Aortopathies.

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  • Additional Information
    • Source:
      Publisher: Wiley-Blackwell Country of Publication: England NLM ID: 101580524 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 2047-9980 (Electronic) Linking ISSN: 20479980 NLM ISO Abbreviation: J Am Heart Assoc Subsets: MEDLINE
    • Publication Information:
      Original Publication: Oxford : Wiley-Blackwell
    • Subject Terms:
      Magnetic Resonance Angiography* ; Computed Tomography Angiography* ; Loeys-Dietz Syndrome*/genetics ; Loeys-Dietz Syndrome*/complications ; Loeys-Dietz Syndrome*/diagnosis ; Loeys-Dietz Syndrome*/diagnostic imaging ; Carotid Arteries*/diagnostic imaging ; Carotid Arteries*/pathology; Humans ; Female ; Male ; Risk Assessment ; Adult ; Middle Aged ; Risk Factors ; Young Adult ; Predictive Value of Tests ; Aortic Dissection/diagnostic imaging ; Aortic Dissection/diagnosis ; Aortic Dissection/surgery ; Vascular Malformations/diagnostic imaging ; Vascular Malformations/diagnosis ; Imaging, Three-Dimensional ; Reproducibility of Results ; Skin Diseases, Genetic/genetics ; Skin Diseases, Genetic/diagnostic imaging ; Skin Diseases, Genetic/diagnosis
    • Abstract:
      Background: Cervical arterial tortuosity is associated with adverse outcomes in Loeys-Dietz syndrome and other heritable aortopathies.
      Methods and Results: A method to assess tortuosity based on curvature of the vessel centerline in 3-dimensional space was developed. We measured cervical carotid tortuosity in 65 patients with Loeys-Dietz syndrome from baseline computed tomography angiogram/magnetic resonance angiogram and all serial images during follow-up. Relations between baseline carotid tortuosity, age, aortic root diameter, and its change over time were compared. Patients with unoperated aortic roots were assessed for clinical end point (type A aortic dissection or aortic root surgery during 4 years of follow-up). Logistic regression was performed to assess the likelihood of clinical end point according to baseline carotid tortuosity. Total absolute curvature at baseline was 11.13±5.76 and was relatively unchanged at 8 to 10 years (fold change: 0.026±0.298, P =1.00), whereas tortuosity index at baseline was 0.262±0.131, with greater variability at 8 to 10 years (fold change: 0.302±0.656, P =0.818). Baseline total absolute curvature correlated with aortic root diameter ( r =0.456, P =0.004) and was independently associated with aortic events during the 4-year follow-up (adjusted odds ratio [OR], 2.64 [95% CI, 1.02-6.85]). Baseline tortuosity index correlated with age ( r =0.532, P <0.001) and was not associated with events (adjusted OR, 1.88 [95% CI, 0.79-4.51]). Finally, baseline total absolute curvature had good discrimination of 4-year outcomes (area under the curve=0.724, P =0.014), which may be prognostic or predictive.
      Conclusions: Here we introduce cervical carotid tortuosity as a promising quantitative biomarker with validated, standardized characteristics. Specifically, we recommend the adoption of a curvature-based measure, total absolute curvature, for early detection or monitoring of disease progression in Loeys-Dietz syndrome.
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    • Contributed Indexing:
      Keywords: LDS; aortic dissection; arterial tortuosity; genetics; thoracic aortic aneurysm
    • Publication Date:
      Date Created: 20240621 Date Completed: 20240702 Latest Revision: 20240720
    • Publication Date:
      20240720
    • Accession Number:
      PMC11255721
    • Accession Number:
      10.1161/JAHA.124.035171
    • Accession Number:
      38904248