A Case of Pan-resistant Burkholderia cepacia Complex Sepsis ... : The Pediatric Infectious Disease Journal (2024)

Division of Pediatric Infectious Diseases, Department of Pediatrics

Division of Neonatology, Department of Pediatrics

Division of Pediatric Infectious Diseases, Department of Pediatrics

Department of Microbiology

Division of Neonatology, Department of Pediatrics Karadeniz Technical University, Faculty of Medicine Trabzon, Turkey

The authors have no funding or conflicts of interest to disclose.

Address for correspondence: Zeynep Gökçe Gayretli Aydin, MD, MSc, Division of Infectious Diseases, Department of Pediatrics, Karadeniz Technical University, Faculty of Medicine, Farabi Hospital, Farabi Street, No: 64, 61080 Trabzon, Turkey. E-mail: [emailprotected].

To the Editors:

B urkholderia cepacia is a Gram-negative, catalase-producing, glucose-nonfermenting, obligately aerobic bacillus distributed widely in water, soil, fruits and vegetables.1 In recent years, B. cepacia has emerged as an important opportunistic pathogen especially in patients with cystic fibrosis, lung transplantation or immunosupression.2 It rarely causes sepsis in neonates in the absence of predisposing factors and can lead to life-threatening infections. Treating B. cepacia poses a challenge due to its intrinsic resistance to aminoglycosides and colistin and becomes even more so when pan resistance develops.3 In this report, we present a case of pan-resistant B. cepacia complex sepsis in a newborn successfully treated with a combination therapy. To the best of our knowledge, this is the first case of a newborn with pan-resistant B. cepacia sepsis that was successfully treated.

The female newborn was referred to our neonatal intensive care unit due to necrotizing enterocolitis (NEC) on the 22nd postnatal day. The newborn was born as a twin via in vitro fertilization during the first pregnancy of a 38-year-old mother, weighed 1240 g at birth and was delivered via cesarean section at 32 weeks. Vital signs were normal at the time of admission. The general condition of the patient was moderate to poor, she appeared icteric. Her capillary refill time was prolonged and her abdomen appeared distended. Given the agents of nosocomial sepsis and NEC, the treatment regimen was maintained with vancomycin and meropenem, and fluconazole was incorporated. Because of cholestasis, meropenem treatment was switched to ceftazidime and amikacin. On the 7th day of hospitalization, her overall condition worsened, accompanied by episodes of low oxygen saturation and apnea attacks. The patient was intubated. B. cepacia was grown in the blood culture. Because the B. cepacia strain exhibited pan resistance, ceftazidime treatment was administered via 3-hour infusion, with the addition of trimethoprim-sulfamethoxazole and ciprofloxacin to the regimen (Table 1). Pan-resistant B. cepacia continued to grow in the blood culture taken at the 72nd hour of treatment. The patient developed thrombocytopenia. Since the patient had NEC and suspicion of a fungal infection, micafungin was commenced. He underwent surgery for NEC and ostomy was performed. Owing to the worsening clinical condition, persistent growth of pan-resistant Burkholderia in the blood culture and regression of cholestasis, ceftazidime was halted, and meropenem infusion was commenced as per the treatment protocol. The patient’s blood culture was sterile on the 7th day of antibiotic therapy. Consequently, meropenem treatment was concluded after a 21-day course, along with ciprofloxacin and trimethoprim-sulfamethoxazole. The patient responded well to this combination therapy. The patient, with stable vitals, maintained normal temperature, achieved successful feeding and weight gain, was discharged on the 41st day of hospitalization.

AKA Amikacin Meropenem İmipenem TMP-SMX Seftazidim Cipro

AKA indicates amoxicillin-clavulanic acid; Cipro, ciprofloxacin; I, intermediate; R, resistant; S, sensitive; TMP-SMX, trimetoprim-sulfamethoxazole.

Antimicrobial resistance remains a significant challenge in nosocomial infections. The emergence of pan-resistant microorganisms over time significantly complicates treatment options, posing formidable challenges for effective management. In this article, we present a case of a newborn infected with pan-resistant B. cepacia, which posed challenges in treatment.

B. cepacia is an opportunistic infectious agent commonly found in individuals with cystic fibrosis and chronic granulomatous disease and those who have undergone organ transplantation.2B. cepacia is an uncommon cause of sepsis in newborns. However, it may lead to infection in newborns, particularly in the presence of underlying risk factors such as prematurity, surgical interventions and instrumentation. The immature immune system due to prematurity and abdominal surgery for NEC in this patient are considered major factors contributing to the development of B. cepacia bacteremia.4

B. cepacia is a microorganism that poses significant challenges in treatment, due to its intrinsic resistance to aminoglycosides and colistin, which can be a potential problem to effective therapy.3 In a study conducted in India, 12 newborns with Burkholderia bloodstream infections were reported. All isolates were sensitive to meropenem and neonates were treated with piperacillin-tazobactam, ciprofloxacin and cotrimoxazole either singly or in combination.4 Studies indicate that Burkholderia can exhibit resistance to every antibiotic by up to 50%. This suggests a high prevalence of multidrug resistance among Burkholderia strains.3,5 In a pediatric study from Turkey, B. cepacia exhibited resistance rates of over 90% to aminoglycosides, 78.2% to piperacillin-tazobactam, 70.8% to meropenem and 84.3% to ciprofloxacin. Additionally, 62.9% of this species was susceptible to trimetoprim-sulfamethoxazole (TMP-SMX), and 75% seemed to be susceptible to levofloxacin.6 In the report by Kim et al,7B. cepacia was 71.4% susceptible to TMP-SMX, 64.3% to ceftazidime, 50% to levofloxacin and 78.6% to meropenem. The Centers for Disease Control and Prevention recommends that treatment decisions should be made on a case-by-case basis, relying on in vitro antibiotic susceptibility data and clinical response. Antibiotic options generally recommended for the treatment of B. cepacia include singly or in combinations containing TMP-SMX, ciprofloxacin, carbapenem or ceftazidime.8 However, no recommendation could be found in the literature regarding the treatment of pan-resistant B. cepacia sepsis. Studies have demonstrated that combination antibiotic therapy with prolonged infusion is effective in eradicating bacteria and reducing mortality rates.9 It is reasonable to employ combination regimens with prolonged infusions to capitalize on antibiotic synergy in severe extensively drug-resistant and pan-drug-resistant infections.10 Therefore, we opted for a combination treatment consisting of meropenem administered via 3-hour infusion, along with ciprofloxacin and TMP-SMX. We achieved a successful response by the end of the treatment. This case represents the first instance in the literature where a newborn with pan-resistant B. cepacia sepsis was successfully treated.


1.Savi D, De Biase RV, Amaddeo A, et al. Burkholderia pyrrocinia in cystic fibrosis lung transplantation: a case report. Transplant Proc. 2014;46:295–297.

2.Woods CW, Bressler AM, LiPuma JJ, et al. Virulence associated with outbreak-related strains of Burkholderia cepacia complex among a cohort of patients with bacteremia. Clin Infect Dis. 2004;38:1243–1250.

3.Avgeri SG, Matthaiou DK, Dimopoulos G, et al. Therapeutic options for Burkholderia cepacia infections beyond co-trimoxazole: a systematic review of the clinical evidence. Int J Antimicrob Agents. 2009;33:394–404.

4.Patra S, Bhat YR, Lewis LE, et al. Burkholderia cepacia sepsis among neonates. Indian J Pediatr. 2014;81:1233–1236.

5.Brady MT, Marcon MJ. Pseudomonas and related genera. In: Cherry JD, Harrison GJ, Kaplan SL, , eds. Textbook of Pediatric Infectious Disease. 7th ed. Sunders Elsevier; 2014:1582–609.

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6.Bedir Demirdag T, Ozkaya Parlakay A, Aygar IS, et al. Major aspects of Burkholderia gladioli and Burkholderia cepacia infections in children. Pediatr Infect Dis J. 2020;39:374–378.

7.Kim KY, Yong D, Lee K, et al. Burkholderia sepsis in children as a hospital-acquired infection. Yonsei Med J. 2016;57:97–102.

8.Centers for Disease Control and Prevention. Burkholderia cepacia in healthcare settings. 2016. Available at: https://www.cdc.gov/hai/organisms/bcepacia.html. Accessed March 24, 2024.

9.Kaur JN, Singh N, Smith NM, et al. Next generation antibiotic combinations to combat pan-drug resistant Klebsiella pneumoniae. Sci Rep. 2024;14:3148.

10.Corona A, De Santis V, Agarossi A, et al. Antibiotic therapy strategies for treating Gram-negative severe infections in the critically ill: a narrative review. Antibiotics (Basel). 2023;12:1262.

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A Case of Pan-resistant Burkholderia cepacia Complex Sepsis ... : The Pediatric Infectious Disease Journal (2024)


What antibiotics is Burkholderia cepacia resistant to? ›

B. cepacia complex strains are intrinsically resistant to a wide range of antimicrobial agents, including aminoglycosides, polymyxin, first and second generation cephalosporins, and carboxypenicillins (51, 54) .

Can you get rid of Burkholderia cepacia? ›

B. cepacia infections need to be treated on a case-by-case basis. These bacteria are often resistant to common antibiotics, but your doctor will likely be able to find some type that will help manage your infection. Laboratory tests can help determine which antibiotics will be most effective in your case.

What are the symptoms of Burkholderia cepacia complex? ›

The symptoms of B. cepacia, if any, are the same as those for any lung infection and can include fever, cough, congestion, shortness of breath, and wheezing.

Should I be worried about Burkholderia? ›

Basic infection prevention and control practices reduce the risk of getting or spreading B. cepacia. These bacteria pose very little medical risk to healthy people.

What is the drug of choice for Burkholderia cepacia? ›

Co-trimoxazole (trimethoprim/sulfamethoxazole) has been a drug of choice.

What kills Burkholderia? ›

Bcc species are intrinsically resistant to many antibiotics such as aminoglycosides and polymyxin B and often require combination therapy to suppress infection in CF. The antibiotics polymyxin, gentamicin and vancomycin are used at high concentrations in B.

Why can't B. cepacia patients get new lungs? ›

Abstract. Background: Pre-operative infection with organisms from the Burkholderia cepacia complex (BCC), particularly B cenocepacia, has been linked with a poorer prognosis after transplantation compared to patients with cystic fibrosis (CF) without this infection.

Why is B. cepacia fatal? ›

Infection by Burkholderia cepacia is sometimes fatal in patients with cystic fibrosis (CF), as the organism can cause necrotising pneumonia and septicaemia (the cepacia syndrome), and is resistant to antibiotics.

How do you get Burkholderia cepacia? ›

It can also be spread by person-to-person contact, contact with contaminated surfaces and exposure to B. cepacia in the environment. B. cepacia usually does not cause illness in healthy people.

What is the old name for Burkholderia cepacia? ›

Burkholderia cepacia (formerly Pseudomonas cepacia) was once thought to be a single bacterial species but has expanded to the Burkholderia cepacia complex (Bcc), comprising 24 closely related opportunistic pathogenic species.

What is the mortality rate for Burkholderia cepacia? ›

Univariate associations of laboratory variables for 14-day and 28-day mortality in patients with Burkholderia cepacia complex. The 14-day mortality (52.1% vs. 0.0%, p<0.001) and 28-day mortality (65.2% vs. 5.5%, p<0.001) were significantly higher in patients in the ICU than those in the general wards.

What precautions should be taken for Burkholderia cepacia? ›

Patient-to-patient spread of B cepacia may be minimized and/or prevented with effective infection-control measures. Use foley catheters only as long as necessary. If possible, avoid their use in compromised hosts predisposed to urinary tract infections (eg, patients with diabetes, SLE, multiple myeloma).

What is the best antibiotic for Burkholderia? ›

Infections due to Bcc can be challenging to manage, as Bcc is intrinsically resistant to a number of commonly used antibiotics. Trimethoprim-sulfamethoxazole (TMP-SMX) and ceftazidime are considered first-line options for Bcc infections (1).

Can you get rid of B cepacia? ›

cepacia is hard to treat and can stay in the body for a long time. It is difficult to treat as it is resistant to many antibiotics. There is evidence that it can become dormant or inactive within the body but then come back.

What is Burkholderia sepsis? ›

Bcc is a rare cause of sepsis in newborns, and its transmission involves human contact with heavily contaminated medical devices and disinfectants. It most commonly presents with respiratory tract, urinary tract, and blood stream infections [4].

What antibiotics are Burkholderia sensitive to? ›

B cepacia often is susceptible to trimethoprim-sulfamethoxazole (TMP-SMX), cefepime, meropenem, minocycline, and tigecycline and has varying susceptibility to ceftazidime and fluoroquinolones.

Is Burkholderia resistant to colistin? ›

However, some Gram-negative species are intrinsically resistant to colistin activity, such as Neisseria meningitides, Burkholderia species, and Proteus mirabilis.

Is Burkholderia cepacia resistant to ceftazidime? ›

Antibiotics to which Burkholderia cepacia has been found sensitive to are ceftazidime (95%), cefotaxime, minocycline, and piperacillin. Antibiotics resistant to Burkholderia cepacia are tetracycline, ticarcillin, and aminoglycosides. In our case, it was resistant to ceftazidime.

What antibiotics are resistant to Burkholderia Pseudomallei? ›

Burkholderia pseudomallei Bp1651 is resistant to several classes of antibiotics that are usually effective for treatment of melioidosis, including tetracyclines, sulfonamides, and β-lactams such as penicillins (amoxicillin-clavulanic acid), cephalosporins (ceftazidime), and carbapenems (imipenem and meropenem).


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