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Paragangliomas (PGLs) are neuroendocrine tumors originating from the neural crest. They usually arise from the adrenal medulla and sympathetic or parasympathetic ganglions. Approximately 10% of PGLs are located in the extra-adrenal gland. Renal PGL is a rare condition. In this case report, we present the 18 F-fluorodeoxyglucose positron emission tomography/computed tomography (PET/CT) and 68 Ga-DOTATATE PET/CT findings of polycystic kidney-derived PGL., Competing Interests: Conflict of Interest: No conflicts of interest were declared by the authors., (Copyright© 2024 The Author. Published by Galenos Publishing House on behalf of the Turkish Society of Nuclear Medicine.)