Unilateral absence of pulmonary artery (UAPA) is a rare congenital anomaly which can seldom be isolated (1:200,000) and incidentally discovered in adulthood. We describe the case of a 54-year-old male patient who was found to have isolated UAPA (IUAPA) during the clinical and radiological investigation of a single episode of hemoptysis. Although abnormal, chest X-ray findings differed considerably from those previously reported and the diagnosis was only achieved by multidetector-row computed tomography angiography and later confirmed by bed-side echocardiography. Further clinical and instrumental investigation revealed systemic hypertension (158/95 mmHg) and bilateral mild hydronephrosis which both remitted after transurethral prostatic adenomyomectomy.
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