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Noncompaction of the ventricular myocardium (NVM) is a rare disease of endomyocardial morphogenesis characterized by numerous,prominent trabeculations and deep intertrabecular recesses.It is commonly associated with congenital heart disease.Isolated noncompaction of the ventricular myocardium (INVM) is thought to be caused by arrest of normal embryogenesis of the endocardium and myocardium.A significant amount of data on INVM are available for the left ventricle but fight ventricular involvement is rare.Although congestive heart failure,arrhythmias,and systemic thromboemboli have been described as three major clinical manifestations of NVM,pulmonary embolism (PE) is an uncommon event.Chronic thromboembolic pulmonary hypertension (CTEPH)caused by isolated noncompaction of the fight ventricular myocardium has never been reported.We describe a case of CTEPH associated with INVM and review the literature to improve the physician's awareness of this disorder.