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.
A 57-year-old man with ADPKD and a limited number of hepatic cysts (Fig. ) was admitted to the hospital because of fever (39.8 °C), right-sided flank pain, diarrhea, vomiting, and headache. Lanreotide injections (120 mg) were started 19 months earlier. At admission, serum CRP was elevated (131 mg/L) and liver enzymes were not elevated (AST 23 U/L, ALT 22 U/L, ALP 71 U/L, GGT 35 U/L). Leukocyturia was excluded. A chest radiograph and abdominal ultrasound were unremarkable. Blood cultures grew E. coli, whereas urine cultures were negative. Under the suspicion of hepatic cyst infection, the patient was hospitalized and treatment with ceftriaxone intravenously was initiated by the local physicians. At discharge, after 3 days, treatment was switched to oral ciprofloxacin for a total of 7 weeks. Recovery was uneventful.\nThe time between injection and development of cyst infection differed widely between patients (1–21 days). The number of days between injection and cyst infection for the individual cases was: 16 (case 1 episode 1) 21 (case 1 episode 2), 8 (case 2), 9 (case 3), 8 (case 4), 16 (case 5), 11 (case 6), and 1 (case 7). It was not documented whether microbiologists were involved in the management of liver cyst infections and whether antibiotic susceptibilities were performed.