A 51-year-old woman with ADPKD and PLD (Fig. ) presented at the hospital because of right upper-quadrant abdominal pain, fever (38 °C), nausea, and vomiting. Lanreotide treatment (120 mg) was started 5 months earlier. At admission, serum CRP was 329 mg/L and serum liver enzymes were normal (AST 29 U/L, ALT 27 U/L, ALP 57 U/L, and GGT 39 U/L). Local physicians initiated piperacillin/tazobactam intravenously under the suspicion of hepatic cyst infection. Blood cultures grew E. coli. Abdominal CT at admission showed no sign of hepatic cyst infection. Patient was hospitalized for 3 days. Treatment was switched to ciprofloxacin twice daily at discharge and continued for 2 weeks. 18F-FDG PET/CT was performed 3 days after discharge and revealed two hepatic cysts showing signs of infection. Recovery was uneventful.
A 46-year old man with ADPKD and PLD (Fig. ) presented with fever (38.7 °C) and complained of acute right upper-quadrant abdominal pain and mild diarrhea (Table ). Lanreotide treatment (120 mg) was started 3 months earlier (Fig. ). At admission, laboratory investigations showed a grossly elevated serum C-reactive protein (CRP, 584 mg/L). Urine sediment examination showed no leukocyturia. Chest radiograph, abdominal ultrasound, and magnetic resonance imaging were unremarkable. Local physicians initiated intravenous antibiotic treatment with three times-daily cefuroxim/metronidazole plus gentamicin bolus injections. The urine culture was negative, but blood cultures grew Escherichia coli. Treatment was switched to oral ciprofloxacin and amoxicillin/clavulanic acid. 18Fluorodeoxyglucose positron-emission computed tomography (18F-FDG PET/CT) was performed at day 8 after admission to locate the infection source, which revealed two hepatic cysts showing signs of infection (Supplementary Fig. 1A). Because of persisting fever, the hepatic cysts were drained in two sessions at day 9. Cyst aspirations did not cause any complications. Both cysts contained pus, but cultures remained negative. After 30 days, the patient was discharged and ciprofloxacin monotherapy was continued for another 4 weeks. Recovery was uneventful. Lanreotide treatment was continued.\nHowever, 4 months after stopping antibiotic treatment, this patient again experienced acute abdominal pain in the right upper quadrant and fever (38.9 °C). Laboratory results showed an increased serum CRP (190 mg/L) and urinalysis excluded leukocyturia. No liver enzyme data were available during this second episode. One month after the diagnosis of the second liver cyst infection episode, Alkaline phosphatase (ALP) and gamma-glutamyl transferase (GGT) were increased (respectively, 97 and 144), aspartate aminotransferase (AST) and alanine aminotransferase (ALT) were normal. Magnetic resonance imaging could not locate the source of infection, and urine and blood cultures were negative. The working diagnosis was recurrent hepatic cyst infection and local physicians started intravenous ceftriaxone treatment, which was switched to oral ciprofloxacin 5 days later. After 6 days, the patient was discharged, and ciprofloxacin was continued for another 15 days. Final recovery was without sequelae. One month after the diagnosis of the second liver cyst infection episode, ALP and GGT were increased (respectively, 97 and 144), as AST and ALT were normal.