A rare case of urinary tract infection by Burkholderia cepacia

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Introduction

B. cepacia was discovered by Walter Burkholder in 1949 as the cause of onion skin rot, and first described as a human pathogen in the 1950s. It was first isolated in patients with cystic fibrosis (CF) in 1977, when it was known as Pseudomonas cepacia. In the 1980s, outbreaks of B. cepacia in individuals with CF were associated with a 35% death rate. B. cepacia has a large genome, containing twice the amount of genetic material as E. coli.

Burkholderia cepacia is a motile, aerobic, non-fermentative, gram-negative bacillus with multi-trichous polar flagella, found in various soil and aquatic environments. It has been widely documented as a lung pathogen in patients with cystic fibrosis and chronic granulomatous disease and is associated with fatal outcomes. Moreover, recently it is documented as an important emerging cause of multi-drug resistant nosocomial infections and consequently, as an important cause of morbidity and mortality [3, 6]. The aim of our study is to present a case of urinary tract infection (UTI) caused by Burkholderia cepacia.

Burkholderia cepacia complex (BCC) is a species complex consisting of Burkholderia cepacia and at least 20 different biochemically similar species of Gram-negative bacteria. They are catalase-producing and lactose-nonfermenting. Members of BCC are opportunistic human pathogens that most often cause pneumonia in immunocompromised individuals with underlying lung disease. Patients with sickle-cell haemoglobinopathies are also at risk.

The group includes B. cepacia, B. multivorans, B. cenocepacia, B. vietnamiensis, B. stabilis, B. ambifaria, B. dolosa, B. anthina, B. pyrrocinia and B. ubonensis, among other species.

Treatment typically includes multiple antibiotics and may include ceftazidime, minocycline, piperacillin, meropenem, chloramphenicol, and trimethoprim/sulfamethoxazole (co-trimoxazole). Although co-trimoxazole has been generally considered the drug of choice for B. cepacia infections, ceftazidime, minocycline, piperacillin, and meropenem are considered to be viable alternative options in cases where co-trimoxazole cannot be administered because of hypersensitivity reactions, intolerance, or resistance. Newer beta-lactam/beta-lactamase combinations like ceftazidime-avibactam or ceftolozane-tazobactam can also be effective. BCC intrinsically resistant to colistin and usually resistant to aminoglycosides.

Case report

A 64-year-old male patient visited the Nikea Primary Healthcare Center, Piraeus, Greece, referred by the family doctor, for follow-up due a history of prostate cancer (patient on immunosuppression) and recurrent UTIs with subsequent admission to the hospital. Patient history revealed diabetes mellitus type 2, arterial hypertension, hypercholesterolemia, hypertriglyceridemia, history of recurrent UTIs, with 4 hospitalizations in a tertiary hospital during the last 2 years, prostatic hypertrophy, 2 episodes of prostatitis before the diagnosis of prostate cancer Gleason score 6, at the end of 2019, with subsequent total prostatectomy, and radiotherapy. Patient history also revealed dysuria, frequent urination, pain and burning sensation during urination and erectile dysfunction. For the recurrent UTIs he had received initially antimicrobial treatment per os, for Pseudomonas spp. There is no further evidence of the antimicrobial treatment he received prior to his admissions to hospital (4 admissions in the last 2 years). On his last admission he received antimicrobial treatment for pseudomonas, his condition improved and he was discharged from the hospital with some of his symptoms persisting, but he does not remember the antimicrobial treatment he received there. However, on worsening of his condition, with subsequent high fever, elevation of CRP, ferritin and ESR values, as well as elevation of leucocyte count, he was admitted to the hospital and received intravenous antimicrobial treatment. The performed urinalysis (Multistix 10 SG Reagent Strips, Siemens Healthineers) and urine culture (incubation at 37°C for 24 hours on MacConkey agar, Columbia blood agar, and Sabouraud dextrose agar for fungi) took place at the Laboratory of Biopathology of Nikea Prime Care Center. We have no information on the diagnosis of the urine culture done at the hospital where the patient was hospitalized.

Results

Urinalysis showed intense pyuria, abundance of micro-organisms and abundance of red blood cells. The urine culture grew monomicrobial Burkholderia cepacia > 10⁵ CFU/ml. The bacterium was identified by the RapID™ REMEL ONE identification system (Thermo Fisher Scientific) (see Figure). Antimicrobial susceptibility testing revealed susceptibility to Ceftazidime, Ciprofloxacin, Norfloxacin, Levofloxacin and Imipenem. The patient was treated with Levofloxacin (3rd generation fluoroquinolone — Tavanic) 500 mg daily per os for 2 weeks, due to his history, and recovered [1].

 

Figure. Identification of Burkholderia cepacia by RapID™ REMEL ONE

 

Discussion

Burkholderia cepacia infections outside the respiratory system are rare. Moreover, recurrent UTIs with B. cepacia is a rare finding, which highlights the importance of our study. UTIs with B. cepacia have been associated with bladder irrigation or use of contaminated hospital objects and liquids [2, 4, 5]. Pseudomonas spp. and Burkholderia spp. have been previously classified in the same genus. Given that they have similar biochemical properties, Burkholderia can be mislabeled as Pseudomonas, as occurred with the patient in our study. B. cepacia is one of the most antimicrobial-resistant organisms and treatment options are limited [2, 4, 5]. The patient does not specify on whose orders he was treated for pseudomonas, at that time he was seen for prostate cancer by a urologist and an oncologist, as well as a diabetologist for diabetes mellitus. In general, this is a patient who has suffered a great deal because of his history.

Conclusion

Burkholderia cepacia is a motile, aerobic, non-fermentative, gram-negative bacillus and has been widely documented as a lung pathogen in patients with cystic fibrosis and chronic granulomatous disease. It is documented as an important emerging cause of multi-drug resistant nosocomial infections, and an important cause of morbidity and mortality.

Diagnosis of infectious diseases, especially in immunodeficiency patients and particularly in urinary tract is of great importance. Early and accurate identification of B. cepacia help patients’ management with proper therapeutic intervention. The critical opinion of doctor microbiologist can help in differential diagnosis among microbes of the same group.

About the authors

C. Seitopoulou

Nikea Prime Care Center

Email: antoniamour@yahoo.com

MD, MSc in Occupational and Enviromental Health, PhDc, Biopathologist, Laboratory of Biopathology

Greece, Nikea

M. Stamouli

Naval and Veterans Hospital of Athens

Email: antoniamour@yahoo.com

BSc, MSc in Health management, MSc in TQM, EurSpLM, Director of Biochemistry Laboratory

Greece, Athens

G. Kalliora

National and Kapodistrian University of Athens

Email: antoniamour@yahoo.com

Student at Faculty of Biology

Greece, Athens

A. Mourtzikou

GHNP “Agios Panteleimon”

Author for correspondence.
Email: antoniamour@yahoo.com

BSc, MSc in Clinical Chemistry, MPH, MPHM, EurSpLM, PhD, Scientific Senior Supervisor, Laboratory of Molecular Diagnostics

Greece, Piraeus

References

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