title: Fatal postoperative systemic pulmonary hypertension in benfluorex-induced valvular heart disease surgery: A case report.
abstract: BACKGROUND
  Drug-induced valvular heart disease (DI-VHD) remains an under-recognized entity.
  
  
  METHODS
  This 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.
  
  
  METHODS
  The diagnostic procedure documented specific drug-induced valvular fibrosis.
  
  
  METHODS
  Surgical mitral and aortic valve replacement was performed.
  
  
  RESULTS
  Heart valve replacement was postoperatively complicated by unanticipated disproportionate pulmonary hypertension. This issue was fatal despite intensive care including prolonged extracorporeal life support.
  
  
  CONCLUSIONS
  Benfluorex 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.
authors.affiliation: 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.
authors.forename: Christophe
authors.initials: C
authors.lastname: Baufreton
authors.forename: Patrick
authors.initials: P
authors.lastname: Bruneval
authors.forename: Marie-Christine
authors.initials: MC
authors.lastname: Rousselet
authors.forename: Pierre-Vladimir
authors.initials: PV
authors.lastname: Ennezat
authors.forename: Olivier
authors.initials: O
authors.lastname: Fouquet
authors.forename: Raphael
authors.initials: R
authors.lastname: Giraud
authors.forename: Carlo
authors.initials: C
authors.lastname: Banfi
mesh_terms: D001067:Appetite Depressants / Q000009:adverse effects*; D017809:Fatal Outcome; D005260:Female; D005277:Fenfluramine / Q000009:adverse effects / Q000031:analogs & derivatives*; D006349:Heart Valve Diseases / Q000139:chemically induced* / Q000150:complications / Q000601:surgery; D019918:Heart Valve Prosthesis Implantation; D006801:Humans; D006976:Hypertension, Pulmonary / Q000209:etiology*; D008875:Middle Aged; D011183:Postoperative Complications / Q000209:etiology*
chemical_list: D001067:Appetite Depressants; D005277:Fenfluramine; C001584:benfluorex