• Christophe Baufreton, Patrick Bruneval, Marie-Christine Rousselet, Pierre-Vladimir Ennezat, Olivier Fouquet, Raphael Giraud, and Carlo Banfi.
• aDepartment of Cardiovascular Surgery, Centre Hospitalier Universitaire d'Angers bDepartment of Pathology, Hôpital Européen Georges Pompidou, Paris Descartes University cDepartment of Pathology, Centre Hospitalier Universitaire d'Angers dDepartment of Cardiology, Centre Hospitalier Universitaire de Grenoble, France eIntensive Care Service, Geneva University Hospitals fHemodynamic Research Group Geneva, Faculty of Medicine, University of Geneva gDivision of Cardiovascular Surgery, Geneva University Hospitals, Geneva, Switzerland.
• Medicine (Baltimore). 2017 Jan 1; 96 (2): e4985.

RationaleDrug-induced valvular heart disease (DI-VHD) remains an under-recognized entity.Patient ConcernsThis report describes a heart valve replacement which was complicated by intractable systemic pulmonary arterial hypertension in a 61-year-old female with severe restrictive mitral and aortic disease. The diagnosis of valvular disease was preceded by a history of unexplained respiratory distress. The patient had been exposed to benfluorex for 6.5 years.DiagnosesThe diagnostic procedure documented specific drug-induced valvular fibrosis.InterventionsSurgical mitral and aortic valve replacement was performed.OutcomesHeart valve replacement was postoperatively complicated by unanticipated disproportionate pulmonary hypertension. This issue was fatal despite intensive care including prolonged extracorporeal life support.LessonsBenfluorex is a fenfluramine derivative which has been marketed between 1976 and 2009. Although norfenfluramine is the common active and toxic metabolite of all fenfluramine derivatives, the valvular and pulmonary arterial toxicity of benfluorex was much less known than that of fenfluramine and dexfenfluramine. The vast majority of benfluorex-induced valvular heart disease remains misdiagnosed as hypothetical rheumatic fever due to similarities between both etiologies. Better recognition of DI-VHD is likely to improve patient outcome.

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