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Fibromuscular Dysplasia (FMD) is a sporadic non-atherosclerotic disease. FMD has been established in nearly every arterial bed. However, the most frequent arteries affected are the renal and carotid arteries. Disease presentation may vary broadly, depending upon the arterial bed complication and the severity of illness. Hypertension, particularly resistant type, headache and dizziness are the most common presentations. String of beads appearance in angiographic views due to post-stenotic aneurysms is the characteristic view. It is most commonly described in young aged females; but in rare male cases has also been reported. Moreover, balloon angioplasty is standard and effective therapy for FMD. We present a young 28-year-old man who was referred for evaluation of resistant hypertension for nearly 3 years without comprehensive workup. The patient underwent renal artery angiography which confirmed beading narrowing of the right renal artery with significant stenosis at mid portion compatible with FMD; and balloon angioplasty was done. This case highlights that FMD should be kept in mind as a rare cause of resistant hypertension in young males; although it is most common in young females.