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CASE REPORT  
Year : 2023  |  Volume : 66  |  Issue : 1  |  Page : 171-173
Burkholderia cepacia causing liver and splenic abscess: Two case reports


1 Department of Medical Gastroenterology, Institute of Medical Sciences and SUM Hospital, Bhubaneswar, Odisha, India
2 Department of Microbiology, Institute of Medical Sciences and SUM Hospital, Bhubaneswar, Odisha, India

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Date of Submission08-Jul-2021
Date of Decision08-Aug-2021
Date of Acceptance20-Nov-2021
Date of Web Publication18-Jan-2023
 

   Abstract 


Burkholderia cepacia infections are common among immunocompromised patients but multiple reports have shown that it can affect immunocompetent patients also. We are reporting two patients with multiple liver and splenic abscesses caused by Burkholderia cepacia. First case is a 54-year-old diabetic male presenting with fever, abdominal pain, bilateral lower limb weakness, and incontinence of urine. Second case is a 41-year-old male presenting with fever and confusion. Both had liver and splenic abscesses. Pus aspirated from the abscesses grew Burkholderia cepacia. Both responded to cotrimoxazole. Our case report emphasizes growing incidence of Burkholderia cepacia in immunocompetent patients.

Keywords: Burkholderia cepacia, liver abscess, splenic abscess

How to cite this article:
Nittala R, Behera MK, Panigrahy R, Narayan J, Mishra D, Singh A, Pati GK, Patil S, Sahu MK. Burkholderia cepacia causing liver and splenic abscess: Two case reports. Indian J Pathol Microbiol 2023;66:171-3

How to cite this URL:
Nittala R, Behera MK, Panigrahy R, Narayan J, Mishra D, Singh A, Pati GK, Patil S, Sahu MK. Burkholderia cepacia causing liver and splenic abscess: Two case reports. Indian J Pathol Microbiol [serial online] 2023 [cited 2023 Feb 8];66:171-3. Available from: https://www.ijpmonline.org/text.asp?2023/66/1/171/367975





   Introduction Top


Burkholderia cepacia (formerly known as Pseudomonas cepacia) is a gram-negative, nonlactose fermenting, aerobic bacilli, which was identified as a plant pathogen more than 50 years ago.[1] It causes opportunistic infection in patients who are suffering from cystic fibrosis and chronic granulomatous infection due to compromised immune function. However, it is now emerging as a multi-drug resistant pathogen even in immunocompetent patients. There are no reports of Burkholderia cepacia causing hepatosplenic abscess in the available literature. We present two case reports of Burkholderia cepacia infection among the immunocompetent patients.

Case 1

A 54-year-old male came with fever with chills since 2 months, abdominal pain since one and half month, weakness of bilateral lower limbs since 6, days and incontinence of urine since 1 day. He was known diabetic on oral hypoglycemic agents. Also, he had COVID-19 infection 2 months prior to these symptoms. He was started empirically on meropenem and other supportive measures. Hemogram showed sepsis with neutrophilia (total leukocyte count was 24 × 103/μl with 93.2% neutrophils and 3.8% lymphocytes; peripheral smear had shown absolute lymphopenia, neutrophilic leukocytosis with toxic changes). Ultrasound abdomen was done which showed multiple microabscesses in both lobes of liver and spleen. The lesion was aspirated from the liver which revealed thick pus [Figure 1]b. Magnetic resonance imaging (MRI) abdomen showed hepatomegaly with multiple clusters of tiny lesions with internal debris in all hepatic segments suggesting abscesses, splenomegaly with multiple tiny T2W hyperintense parenchymal lesions with diffusion restriction [Figure 1]a, and features of evolving microabscess in right kidney. Also, high resolution computed tomography (CT) thorax had shown pulmonary septic emboli [Figure 1]c and [Figure 1]d. The blood sample was collected in a BACT/ALERT FA PLUS, BIOMÉRIEUX blood culture bottle. Then the flagged positive culture bottle was inoculated into blood agar and MacConkey agar. After 18–24 h of aerobic incubation at 37°C, nonlactose fermenting colonies were grown. These were gram-negative motile rods with positive oxidase and catalase reaction. Then isolated colony was put in VITEK® 2 AST N281 card and the organism was found to be Burkholderia capacia in VITEK 2 automated culture system [Figure 1]e. The organism was found to be resistant to polymixin B and colistin and susceptible to cotrimoxazole. The patient was started on cotrimoxazole. After 3 days, he became afebrile and his total leukocyte counts started decreasing. After the total leukocytes came to normal range, intravenous methylprednisolone 1 g intravenously was given for 5 days for myelitis. He was shifted to intravenous insulin due to steroid-induced hyperglycemia. His power in the lower limbs improved, he was able to walk normally, and was able to pass urine normally.
Figure 1: a: MRI abdomen showing multiple liver and splenic abscesses (red arrows). It shows septated cystic lesions with internal debris and multiple tiny T2W hyperintense parenchymal lesions with diffusion restriction. b: MRI of cervical spine showing subtle intramedullary high cord signal opposite C4 to C6 vertebrae (myelitis). c & d: High resolution computed tomography of thorax showing septic pulmonary emboli with air-fluid levels. e: Culture media of Burkholderia cepacia; Source-Department of Radiodiagnosis, Department of Microbiology, IMS & SUM Hospital

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Case 2

A 41-year-old male presented with high-grade intermittent fever with chills and rigor for 3 months and confusion for 5 days. On examination, he had hepatosplenomegaly. No neck rigidity was present. He was admitted in intensive care unit. He was started empirically on intravenous antibiotics. His blood reports showed raised total leucocyte count with a decrease in platelet count. Liver function tests and renal function tests were normal. Serum electrolytes were normal. Imaging of brain was done, it was normal. Ultrasound abdomen showed hepatosplenomegaly with multiple small cystic lesions scattered in the parenchyma of liver and spleen. Pus was aspirated from these cystic lesions and sent for microbiological examination. Gram stain of the sample showed plenty of gram-negative rods with pus cell. Then the sample was inoculated into blood agar and MacConkey agar. After 18–24 h of aerobic incubation at 37°C, the nonlactose fermenting colonies were processed as described in case number 1. Intravenous cotrimoxazole was started and his sensorium improved and fever spikes subsided. It was given for 2 weeks and the patient was discharged. On follow-up, ultrasound of the abdomen was reviewed, which showed resolution of the previous abscess.


   Discussion Top


B. cepacia can present multi-organ dysfunction syndrome, psoas abscess, pyopneumothorax, and bacteremia in immunocompetent patients as reported in previous literature.[1],[2],[3],[4] B. cepacia has been found to contaminate antiseptics, disinfectants, nebuliser solution, and dextrose solution, posing a great threat to inpatients. It has also been found to cross-infect between cystic fibrosis patients and noncystic fibrosis patients. B. cepacia has emerged as an important cause of morbidity and mortality in hospitalized patients, particularly in intensive care units (ICUs) and cancer centers, largely because of high intrinsic antibiotic resistance.[5]

Liver abscess is a common entity in India; simultaneous presence of liver and splenic abscess are rare presentations observed in clinical practice. Splenic abscess is a rare entity with a high mortality if left untreated.[6] It is caused by bacterial, fungal, mycobacterial organisms and commonly seen in immunocompromised patients suffering from immunodeficiency syndromes, cancers, etc. It has also been seen in Crohn's disease and ulcerative colitis. Causes of multiple splenic abscess have been bacterial and fungal including Salmonella typhi, Staphylococcus aureus, Klebsiella pneumonia, Mycobacterium avian complex, Enterococcus spp., Candida parapsilosis, Streptococcus intermedius, Burkholderia pseudomallei, and Propionibacterium acnes. Burkholderia pseudomallei is known to cause isolated and multiple liver abscess, multiple splenic abscess, and also combined liver and splenic abscess leading to melidiosis.[7],[8] Burkholderia cepacia is known to cause multiple liver and splenic abscess separately.[5] But our patients had both liver and splenic abscesses. The risk factor was type 2 diabetes mellitus in one of our case and also he had prior COVID-19 infection 3 months back. Our second patient had no risk factors and no prior COVID-19 infection. There are no previous reports of myelopathy and pulmonary septic emboli in patients infected with Burkholderia cepacia, as reported in our case. There has been a report of brain abscess due to Burkholderia cepacia.[9] Both of our patients did not respond to empirically given third-generation cephalosporins, but responded very well to combination of trimethoprim-sulphmethoxazole. The growing incidence of Burkholderia cepacia in immunocompetent patients and its atypical presentations should be kept in mind to avoid delay in diagnosis and initiate early treatment.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
McClean S, Callaghan M. Burkholderia cepacia complex: Epithelial cell-pathogen confrontations and potential for therapeutic intervention. J Med Microbiol 2009;58:1-12.  Back to cited text no. 1
    
2.
Karanth SS, Regunath H, Chawla K, Prabhu M. A rare case of community acquired Burkholderia cepacia infection presenting as pyopneumothorax in an immunocompetent individual. Asian Pac J Trop Biomed 2012;2:166-8.  Back to cited text no. 2
    
3.
Ranjan R, Chowdhary P, Kamra A. Community acquired Burkholderia cepacia bacteraemia presenting as MODS in an immunocompetent individual: An unusual case. J Clin Diagn Res 2017;11:DD01-2.  Back to cited text no. 3
    
4.
Agrawal S, Kapil A, Dhawan B, Sharma BS. Burkholderia cepacia: An uncommon cause of bilateral primary psoas abscesses in a patient with a Pott spine that cannot be ignored. Natl Med J India 2018;31:124-5.  Back to cited text no. 4
  [Full text]  
5.
Mukhopadhyay C, Bhargava A, Ayyagari A. Two novel clinical presentations of Burkholderia cepacia infection. J Clin Microbiol 2004;42:3904-5.  Back to cited text no. 5
    
6.
Ng KK, Lee TY, Wan YL, Tan CF, Lui KW, Cheung YC, et al. Splenic abscess: Diagnosis and management. Hepatogastroenterology 2002;49:567-71.  Back to cited text no. 6
    
7.
Lee YL, Lee SS, Tsai HC, Chen YS, Wann SR, Kao CH, et al. Pyogenic liver abscess caused by Burkholderia pseudomallei in Taiwan. J Formos Med Assoc 2006;105:689-93.  Back to cited text no. 7
    
8.
Pal P, Ray S, Moulick A, Dey S, Jana A, Banerjee K. Liver abscess caused by Burkholderia pseudomallei in a young man: A case report and review of literature. World J Clin Cases 2014;2:604-7.  Back to cited text no. 8
    
9.
Hobson R, Gould I, Govan J. Burkholderia (Pseudomonas) cepacia as a cause of brain abscesses secondary to chronic suppurative otitis media. Eur J Clin Microbiol Infect Dis 1995;14:908-11.  Back to cited text no. 9
    

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Correspondence Address:
Manas K Behera
SOA Institute of Medical Sciences and SUM Hospital, K8 Lane 1, Kalinga Nagar, Bhubaneswar, Odisha - 751 003
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpm.ijpm_702_21

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