Background: Cowper's syringocele is a rare condition, uncommon in the adult population. It is more common in the pediatric population. Patients could be asymptomatic or present with irritative and/or obstructive symptoms. Ultrasonography (US) of perineal region, pelvis MRI, or urethrography are the most often used diagnostic modalities. Surgical treatment is indicated in all symptomatic patients. Endoscopic approach is preferred over open surgery. Unroofing of syringocele with cold knife or holmium yttrium aluminium garnet (YAG) laser is effective treatment. Case Presentation: A 17-year-old Caucasian male presented at our emergency department with irritative symptoms. US of perineal region showed cystic formation near urethra. Pelvis MRI showed Cowper's gland syringocele. Endoscopic unroofing with holmium YAG laser was performed. Six months postoperatively the patient was asymptomatic and satisfied with the treatment. Conclusion: Cowper's syringocele is a rare condition. Urologist should consider this condition in pediatric and adolescent patients with irritative and/or obstructive symptoms. Pelvis MRI and perineum US are the main diagnostic tools. Surgical treatment is effective. Endoscopic approach is preferred. Open surgery is indicated in complicated cases and in cases when endoscopic treatment fails.