A 54-year-old woman with ADPKD and PLD (Fig. ) presented with fever (39.6 °C) and cold shivers without abdominal pain, after having experienced abdominal complaints during a few weeks following each lanreotide injection. Lanreotide treatment (120 mg) was started 5 months earlier. At admission, serum CRP was 245 mg/L and some liver enzymes were elevated (ALT 62 U/L, ALP 119 U/L, and GGT 83 U/L). Leukocyturia was excluded with urine sediment analysis. Abdominal ultrasound showed no abnormalities, except for gallbladder sludge. Blood cultures grew E. coli. The patient was hospitalized for 3 days and treated by local physicians under the suspicion of hepatic cyst infection with cefuroxime intravenously. At discharge, treatment was switched to ciprofloxacin for another 11 days. 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.