Treatment preferences of adult patients with hereditary fructose intolerance: A discrete choice experiment

Abstract

Background: Patients with hereditary fructose intolerance (HFI) follow a fructose-restricted diet to avoid life-threatening complications. The aim of this study was to investigate whether patients with HFI have a preference for a pharmacological therapy as an add-on or replacement of their current diet.

Methods: Adult patients with HFI recruited from metabolic clinics and through social media (n = 90) were asked to complete a labelled discrete choice experiment (DCE). The DCE was composed after personal interviews with patients (n = 3) and health care providers (n = 6), and contained 12 choice sets. In each choice set, patients were asked to choose between their current fructose-restricted diet or pharmacological therapy, which consisted of four attributes: side effects, costs, mode of administration and effect on fructose restriction.

Results: Although the random parameter model showed that patients on average preferred their current diet over a putative pharmacological therapy, 86 % of the patients opted for pharmacological therapy in at least one of the choice tasks. One tablet daily on special occasions without any fructose restriction, no side effects and no additional costs were significantly associated with a preference for pharmacological therapy. Younger age, more daily impact of the disease and more daily impact of the dietary restriction were associated with a preference for pharmacological therapy.

Conclusions: Under specific conditions, patients with HFI prefer a pharmacological therapy over their fructose-restricted diet. Therapy-related factors as well as patient-related factors play a role in this choice. These findings may guide the development and implementation of a pharmacologic therapy for patients with HFI.

Keywords: Discrete choice experiment; Disease burden; Fructosemia; HFI; Pharmacological therapy; Quality of life.