BackgroundCyst infections is not common in the patients with autosomal dominant polycystic kidney disease (ADPKD) however it may pose major problems to the clinicians because the diagnosis is hampered by lack of reliable imaging techniques for identification of the infected cysts and treatment may be difficult due to poor penetration of antibiotics into the cysts.Case presentationWe present a case of ADPKD and intractable pyocysts that did not respond to standard antibiotic therapy but successfully treated by using ultrasound-guided cyst puncture, and repeated irrigation and drainage.ConclusionWhere the experienced interventional radiologists are available, this method can rescue these patients from nephrectomy.