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Isolated bicuspid pulmonary valve is a rare arterial valve anomaly with very few reports in the literature, which regard overall cases of postmortem diagnosis. However, the true incidence of bicuspid pulmonary valve could be underestimated because of the difficulty in imaging pulmonary valve morphology with conventional two-dimensional transthoracic echocardiography (TTE). We report a case of isolated bicuspid pulmonary valve in an adult diagnosed by three-dimensional TTE. Off line multidimensional analysis of full-volume 3D allowed to obtain a short axis view of pulmonary valve showing a bicuspid valve characterized by an anterior and a posterior cups, normally thickened and with preserved systolic opening.