A 22-year-old man has consulted in emergency for acute urinary retention and left renal colic. Bladder catheterization was performed. Symptomatic treatment was provided with no improvement. MRI showed a pseudotumoral bladder wall thickening associated with vesical floor budding with prostate median lobe infiltration. The patient got an endoscopy that concluded to an inflammatory aspect of the bladder mycosa and a solid mass in the bladder neck arising. The biopsy during examination concluded to a glandular cystitis. Ultrasonography performed six months later still showed an enlarged prostate of 60g volume, post void residue of 280ml and bilateral hydronephrosis. A second cystoscopy showed an obstructive prostate with a median lobe. A transurethral resection of this lobe was performed. The pathological examination concluded to a benign prostate hyperplasia. This case is likely to be the first reported so far about a BPH in a young male associated with Cystitis Glandularis. Neither etiology nor evidence of the cause behind this case has been identified so far. Although Benign Prostate Hypertrophia is rare among young males, its ethiopathogenesis is not well known, its relation with cystitis glanduralis in young patients has never been described before. Both medical and surgical approaches remain similar to the adults.