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Arterial tortuosity syndrome (ATS) is a very rare autosomal recessive disease which results from mutations of the SLC2A10 gene. In this article, we describe the results of vascular imaging of an adult patient with ATS who was presented with pulmonary hypertension and after a complete evaluation, he was a candidate for interventional balloon angioplasty. Our patient is one of the very limited numbers of list patients in the studies for whom percutaneous balloon angioplasty performed successfully.