Abstract
ObjectivesTo inform the design of a clinical trial of a targeted screening programme for relatives of individuals affected by thoracic aortic disease, we performed a consensus exercise as to the acceptability of screening, the optimal sequence and choice of tests, long-term patient management, and choice of trial design.MethodsWorking with the Aortic Dissection Awareness UK & Ireland patient association, we performed a Delphi exercise with clinical experts, patients, and carers, consisting of three rounds of consultation followed by a final multi-stakeholder face-to-face workshop.ResultsThirty-five experts and 84 members of the public took part in the surveys, with 164 patients and clinicians attending the final workshop. There was substantial agreement on the need for a targeted screening pathway that would employ a combined approach (imaging + genetic testing). The target population would include the first- and second-degree adult (> 15 years) relatives, with no upper age limit of affected patients. Disagreement persisted about the screening process, sequence, personnel, the imaging method to adopt, computed tomography (CT) scan vs magnetic resonance imaging (MRI), and the specifics of a potential trial, including willingness to undergo randomisation, and measures of effectiveness and acceptability.ConclusionA Delphi process, initiated by patients, identified areas of uncertainty with respect to behaviour, process, and the design of a targeted screening programme for thoracic aortic disease that requires further research prior to any future trial.
Highlights
Thoracic aortic disease is an uncommon condition that remains asymptomatic for many years until it presents as an acute aortic syndrome
The long latency period, the ready availability of cross sectional imaging, and the emergence of high through-put genetic testing all support the introduction of targeted screening in people at risk of non-syndromic aortic disease
It is reasonable to assume that high-risk non-syndromic phenotypes will benefit from intensive surveillance, strict blood pressure control, and early surgery
Summary
Thoracic aortic disease is an uncommon condition that remains asymptomatic for many years until it presents as an acute aortic syndrome These are often fatal, accounting for up to 5000 emergency hospital admissions and approximately 2000 deaths per year in the UK [1,2,3], more. The optimal design of an effective screening programme is not specified To address this uncertainty, and as part of a patient-led initiative to reduce unwanted variation in the care of people at risk of death from aortic disease, we performed a Delphi exercise to identify the scope of a future research programme that would evaluate the acceptability, clinical effectiveness, and cost effectiveness of a targeted screening programme in at risk-populations
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