• Chest · Oct 2011

    Case Reports

    Pulmonary arterial hypertension in a patient with Cowden syndrome and anorexigen exposure.

    • Delphine Natali, Barbara Girerd, David Montani, Florent Soubrier, Gérald Simonneau, Marc Humbert, and Olivier Sitbon.
    • Faculté de Médecine, Université Paris-Sud, Kremlin-Bicêtre, France.
    • Chest. 2011 Oct 1;140(4):1066-8.

    AbstractWe report a case of pulmonary arterial hypertension (PAH) occurring in a patient with Cowden syndrome with a mutation in the phosphatase and tensin (PTEN) tumor suppressor gene, in the context of exposure to the appetite suppressant dexfenfluramine. Anorexigen exposure is known to be a risk factor for PAH. However, the role of PTEN in cell function and the development of pulmonary vascular remodeling and histopathologic signs of PAH in mice with a Pten depletion in smooth muscle cells suggest that the association of PAH and Cowden syndrome may be relevant. In this case report, we hypothesize that PTEN mutations may be a predisposing factor for the development of PAH, with anorexigen exposure as a potential trigger.

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