J Infect Chemother (2013) 19:969–971 DOI 10.1007/s10156-012-0547-8
CASE REPORT
Burkholderia contaminans: unusual cause of biliary sepsis Goh Ohji • Kiyofumi Ohkusu • Akihiro Toguchi Yoshihito Otsuka • Naoto Hosokawa • Kentaro Iwata
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Received: 9 August 2012 / Accepted: 26 December 2012 / Published online: 6 January 2013 Ó Japanese Society of Chemotherapy and The Japanese Association for Infectious Diseases 2013
Abstract We report a case of biliary tract infection caused by a strain of Burkholderia contaminans, a member of the Burkholderia cepacia complex. The patient developed sepsis after endoscopic retrograde cholangiopancreatography (ERCP). Gram-negative bacilli were isolated from blood and bile cultures. Automated bacterial identification systems identified the organism as Burkholderia cepacia, whereas DNA sequence analysis revealed that the recA gene isolate was identical to that of B. contaminans. The patient responded to therapy with the antibiotics trimethoprim/sulfamethoxazole and biliary tract decompression. This case suggests that B. contaminans can be a
G. Ohji (&) K. Iwata Division of Infectious Disease Therapeutics, Department of Microbiology and Infectious Diseases, Kobe University Graduate School of Medicine, 7-5-2 Kusunoki-cho, Chuo-ku, Kobe, Hyogo 650-0017, Japan e-mail:
[email protected] G. Ohji K. Iwata Research Center for Urban Safety and Security, Kobe University, Kobe, Japan G. Ohji Department of Clinical Laboratory, Kobe University Hospital, Kobe, Japan K. Ohkusu Department of Microbiology, Gifu University Graduate School of Medicine, Gifu, Japan A. Toguchi Y. Otsuka N. Hosokawa Department of Laboratory, Kameda Medical Center, Kamogawa, Japan N. Hosokawa Department of General Medicine and Infectious Diseases, Kameda Medical Center, Kamogawa, Japan
causative agent of healthcare-associated biliary tract infections such as ERCP-related cholangitis. Keywords Burkholderia contaminans Burkholderia cepacia complex ERCP Biliary tract infection Cholangitis
Introduction Burkholderia cepacia complex (Bcc) is a group of glucose-nonfermentative, gram-negative, and ubiquitously distributed aerobic bacilli. Bcc can cause healthcareassociated infections such as pneumonia, central-lineassociated bloodstream infection (CLABSI), and urinary tract infection (UTI) in patients who have a history of broad-spectrum antimicrobial agent administration [1]. Burkholderia contaminans was previously described as a member of the Bcc taxon K complex [2]. Similar to other Bcc species, B. contaminans can cause healthcareassociated infections [3].
Case report A previously healthy 57-year-old woman was admitted to our tertiary care hospital because of progressive jaundice. Physical examination revealed no tenderness in the epigastrium. Abdominal ultrasonography revealed intrahepatic and common bile duct dilatation. The patient’s hematological data were as follows: white blood cell (WBC) count, 5,700/ mm3; aspartate aminotransferase (AST) level, 61 IU/l; alanine transaminase (ALT) level, 99 IU/l; total bilirubin, 9.1 mg/dl; and direct bilirubin, 7.1 mg/dl. The patient was suspected of having bile duct obstruction caused by a tumor.
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On the third day of hospitalization, endoscopic retrograde cholangiopancreatography (ERCP) was performed, which revealed distal bile duct stricture by the tumor. An internal stent was inserted for drainage, and bile was collected during ERCP for microbiological diagnosis. The bile cultures showed no bacterial growth. Prophylactic antibiotics were not administered before ERCP. On the fifth day of hospitalization, the patient became febrile (39.2 °C) and showed tachycardia (105/min), tachypnea (32/min), and leukocytosis (12,400/mm3); the total bilirubin remained elevated (4.3 mg/dl). Sepsis and cholangitis caused by stent occlusion were suspected. Emergency ERCP was performed to replace the internal stent, and stent occlusion was confirmed. Simultaneously, bile and blood samples were obtained for culture. The patient was then provided empiric treatment with the antibiotics cefoperazone-sulbactam. On the seventh day of hospitalization, blood culture showed gram-negative bacilli. An aliquot was obtained from a positive blood culture bottle, inoculated in trypticase soy broth and 5 % sheep blood agar, and cultured aerobically at 37 °C. After 6 h, smooth colonies appeared and induced hemolysis on the blood agar. The bile sample was also aerobically cultured on 5 % sheep blood agar at 37 °C. This culture also yielded smooth colonies that induced hemolysis after 24 h. Isolates from both cultures were identified as B. cepacia by using Microscan Walkaway (Siemens Healthcare Diagnostics, USA) and RapID NF plus identification strips (Remel, USA). Sensitivity tests were performed using Microscan Walkaway and microdilution methods as per the Clinical Laboratory Standards Institute (CLSI) guidelines. Both isolates were resistant to aminoglycoside, fluoroquinolone, and b-lactam antibiotics, except carbapenem, and were susceptible to trimethoprim-sulfamethoxazole and minocycline. Subsequently, the antibiotics were changed to trimethoprim-sulfamethoxazole to target the causative organism. The patient became afebrile the following day, and the total bilirubin level decreased to 1.2 mg/dl on the 13th day of hospitalization. The antibiotic treatment was continued for 14 days. Complications of cholangitis and recurrence of bacteremia have not been observed subsequently. Commercial test systems have been reported to lack sufficient specificity to identify B. cepacia complex isolates [4, 5]. Hence, DNA sequence analysis was used to identify this isolate. The recA gene was amplified using primers BCR1 and BCR2 as described by Mahenthiralingam et al. [6]. This analysis revealed that the recA gene sequence of the isolate was 99 % identical to that of B. contaminans [2].
Discussion In our patient, blood culture analysis revealed B. contaminans. The bile culture collected during ERCP cannulation also
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yielded B. contaminans. Because the patient’s urine culture was negative, and she had undergone neither central venous catheter nor peripheral venous catheter insertion, other causes of bacteremia such as CLABSI or UTI were ruled out, and B. contaminans was confirmed to be the causative organism of bacteremic cholangitis. Gram-negative bacilli such as Escherichia coli, Klebsiella spp., and Enterobacter spp. have been usually reported to be the causative organisms of acute cholangitis, although anaerobes have also been suggested as the causative organisms [7, 8]. To our knowledge, only one case of cholangitis caused by Burkholderia spp. has been reported in the English literature [9]. Lai et al. [9] reported acute cholangitis caused by B. pseudomallei. Several species of the Burkholderia genus, including Bcc strains, B. pseudomallei, and B. mallei, are known to cause infection in humans. However, the clinical presentation, transmission mode, and epidemiology of each Burkholderia species differ markedly [1, 10]. Clinical presentations of B. pseudomallei infection, melioidosis, are pneumonia, skin lesions, or multiorgan abscess secondary to bacteremia, whereas those of Bcc infections are healthcare-associated infections such as CLABSI, UTI, or pneumonia [10]. Melioidosis is an endemic disease mainly in Southeast Asia and acquired from soil or surface water in farms, whereas Bcc infections are not an endemic disease and are acquired mainly via healthcare settings [1, 10]. Lai et al. [9] reported B. pseudomallei-induced cholangitis complicated with pneumonia and septic shock in an endemic area. Our case was healthcare-associated cholangitis secondary to ERCP. This finding suggests that although B. pseudomallei and Bcc strains belong to the same genus, Burkholderia, their clinical implications in biliary tract infection differ. Because there are no reports of B. contaminans and other Bcc organisms as the causative agents of cholangitis, our case, to our knowledge, is the first known biliary tract infection caused by Bcc. Infection is a morbid complication of ERCP and can be associated with ERCP-related death. The most frequent septic complication of ERCP is cholangitis, which results from use of a contaminated device or incomplete drainage [11]. Acute cholangitis in our patient was caused by stent occlusion after drainage. Because bile culture was positive after the second ERCP, it might be a case of device-related cholangitis, similar to a previously reported case [9]. We surveyed other endoscopy-related infections in our hospital; however, no other such cases were identified. We also checked the decontamination procedures of the endoscope and confirmed that all the procedures conformed to the guidelines of the Japan Gastroenterological Endoscopy Society. As no other endoscopy-related infections were identified, we did not perform environmental cultures. Furthermore, hygiene management and aseptic techniques were confirmed in our Endoscopy Department after the detection of device-related cholangitis in our case.
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The American Society for Gastrointestinal Endoscopy recommends antibiotic prophylaxis before ERCP in patients who do not achieve complete drainage for known or suspected biliary obstructions [12]. We expected complete drainage by internal stenting in our patient and did not administer prophylactic antibiotics. One randomized controlled trial suggested beneficial effects of continued antibiotic administration until complete biliary drainage in patients who undergo an ERCP for biliary obstruction [13]. However, antibiotic prophylaxis administration and its continuation after the first ERCP with routine antibiotics such as gentamicin, ciprofloxacin, and b-lactam [14] would have been ineffective in this case because these antibiotics have no in vitro activity against B. contaminans. Decontamination of endoscopes and adequate aseptic techniques during ERCP might be essential to prevent endoscopyrelated Bcc infections. Proposing a therapeutic recommendation would be difficult because of the lack of reports of biliary tract infections caused by Burkholderia spp. In our case, trimethoprimsulfamethoxazole was administered for 2 weeks. Meropenem, chloramphenicol, and minocycline are alternative effective agents against Bcc [1]. The isolates in our case were susceptible to meropenem and minocycline and hence could have been used as alternative agents to trimethoprimsulfamethoxazole. Because neither recurrent bacteremia nor cholangitis occurred, the treatment might be sufficient, at least in this case. Further studies are required to determine antibiotic treatments for biliary tract infections caused by Bcc. In summary, we report a first known case of bacteremic biliary tract infection caused by B. contaminans following ERCP. B. contaminans can be a causative organism of healthcare-associated biliary tract infections. Conflict of interest
None to declare.
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