Fatal Infection Caused by Cupriavidus gilardii in a Child with Aplastic Anemia

Abstract

Cupriavidus gilardii is a Gram-negative bacterium that has rarely been associated with human infections. We report a fatal case of sepsis caused by C. gilardii in a previously healthy 12-year-old female. CASE REPORTWhile vacationing in Europe, a previously healthy 12-yearold female was diagnosed with severe idiopathic aplastic anemia. One week after presentation, the patient was transferred to a hospital in the United States near her home. Laboratory evaluation at the time of admission was significant for a total white blood cell count of 2,590/l with a differential of 98.8% lymphocytes, 0.4% monocytes, and 0.8% neutrophils, hemoglobin of 7 g/dl, and platelets of 72,000/l. On hospital day (HD) 1, following placement of a double-lumen Hickman catheter, the patient became febrile to 39.7°C and was started on piperacillin-tazobactam. She remained febrile through HD 12, prompting empirical addition of amikacin and then liposomal amphotericin B to her antibiotic regimen. A contrast computed tomography (CT) scan of the chest, abdomen, and pelvis showed diffuse perirectal thickening, prompting initiation of metronidazole for improved anaerobic coverage. She subsequently defervesced and remained afebrile from HD 15 to 24, though all antimicrobials were continued due to profound neutropenia and persistent perirectal pain.A repeat CT scan on HD 25 revealed progression of moderate to marked perirectal inflammation associated with the appearance of new fluid collection not amenable to surgical drainage. On HD 27, a blood culture from the previous day became positive for vancomycin-resistant enterococcus (VRE), and her central line was removed after initiation of linezolid. By HD 30, persistent fevers and perirectal pain resulted in discontinuation of piperacillin-tazobactam in favor of meropenem, and from HD 32 to 38, the patient clinically improved. During this time, amikacin and liposomal amphotericin B were discontinued, and central venous access was reestablished with a peripherally inserted catheter (PICC line).Low-grade fever (38.0 to 38.3°C) and abdominal pain recurred on HD 40 and persisted through HD 53, when the patient developed high spiking fevers. A repeat CT scan revealed worsening of perirectal inflammation and progression of bowel wall inflammation to involve the cecum, most of the ascending colon, and a portion of the transverse colon. Stool testing for Clostridium difficile antigen was negative. However, a stool surveillance culture was positive for Cupriavidus gilardii, resistant to piperacillin-tazobactam, aztreonam, imipenem, and meropenem, and susceptible to cefepime, trimethoprim-sulfamethoxazole, and ciprofloxacin. Consequently, meropenem was discontinued in favor of cefepime, and amikacin was added. Fever subsided, and linezolid was discontinued. On HD 60, the patient became febrile again, and blood cultures were positive for VRE and C. gilardii, now susceptible only to trimethoprim-sulfamethoxazole and ciprofloxacin.Ciprofloxacin and linezolid were added, and after removal of the PICC line, the pat...

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