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  Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 38  |  Issue : 2  |  Page : 222-225
 

Burkholderia pseudomallei septic arthritis in Type-2 diabetes mellitus patients: Report of two cases


1 Department of Microbiology, Excelcare Hospitals, Guwahati, Assam, India
2 Scientist-E and Nodal Officer, VRDL, ICMR RMRC, Dibrugarh, Assam, India
3 Department of Internal Medicine, Excelcare Hospitals, Guwahati, Assam, India
4 Department of Endocrinology, Excelcare Hospitals, Guwahati, Assam, India
5 Scientist D, ICMR RMRC, Dibrugarh, Assam, India

Date of Submission21-Feb-2020
Date of Decision14-Jun-2020
Date of Acceptance26-Jun-2020
Date of Web Publication29-Aug-2020

Correspondence Address:
Dr. K Baruah Frincy
Excel care Hospitals, Guwahati, Assam
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijmm.IJMM_20_74

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 ~ Abstract 


Two cases of Burkholderia pseudomallei septic arthritis are presented with a brief review of the literature. B. pseudomallei septic arthritis most commonly occurs in diabetics and other immunocompromised patients and may prove fatal despite appropriate therapy. Clinical and microbiological suspicion of B. pseudomallei infection may help in providing appropriate empirical therapy.


Keywords: Burkholderia pseudomallei, diabetes mellitus, septic arthritis


How to cite this article:
Frincy K B, Biswajyoti B, Saikia S, Baruah MP, Devi U. Burkholderia pseudomallei septic arthritis in Type-2 diabetes mellitus patients: Report of two cases. Indian J Med Microbiol 2020;38:222-5

How to cite this URL:
Frincy K B, Biswajyoti B, Saikia S, Baruah MP, Devi U. Burkholderia pseudomallei septic arthritis in Type-2 diabetes mellitus patients: Report of two cases. Indian J Med Microbiol [serial online] 2020 [cited 2020 Oct 24];38:222-5. Available from: https://www.ijmm.org/text.asp?2020/38/2/222/293913





 ~ Introduction Top


Burkholderia pseudomallei infection may present as septicaemia with abscess, osteomyelitis or septic arthritis. Diabetes mellitus, renal disease, alcoholism, liver cirrhosis, thalassaemia, etc., are predisposing conditions. Here, we report two fatal cases of B. pseudomallei septic arthritis in patients of type 2 diabetes mellitus (T2DM) despite appropriate treatment. Appropriate written informed consents were obtained from the patients for reporting these findings for publication.


 ~ Case Reports Top


Case 1

A 72-year-old male was admitted with chief complaints of fever with chills and rigors, altered mental status and severe pain in the right ankle. He had been suffering from pain and swelling in the right ankle joint following an accidental fall from bed 10 days back. The patient was a farmer by profession with a known history of hypertension for 14 years and T2DM for 7 years. He was on insulin therapy for T2DM. On examination, there was tenderness and swelling of the right ankle. A diagnosis of septic arthritis was performed, and meropenem and linezolid were started.

On the day of admission, his temperature was 103°F, blood pressure (BP): 180/80 mm Hg, heart rate (HR): 106/min and respiratory rate: 20/min. Random blood sugar was 324 mg/dl. Haematological findings were as follows: haemoglobin (Hb): 9.8 mg/dl, total leucocyte count (TLC): 7.3 × 103/μL, erythrocyte sedimentation rate (ESR): 45 mm/h, C-reactive protein (CRP): 164.2 mg/L. X-ray (anteroposterior and lateral view) of the right ankle revealed only soft-tissue swelling around the ankle joint, while the musculoskeletal ultrasonography (USG) revealed right ankle joint effusion, synovitis of tibialis anterior tendon, retrocalcaneal bursitis with inflamed Kager fat and diffuse cellulitis. Arthrotomy was done for the evacuation of pus and the sample was sent to the Microbiology department for culture and sensitivity. Sample processing was done as per the standard protocol. Gram stain of the pus showed numerous pus cells and Gram-negative bacilli with typical bipolar staining. After 24 h of aerobic incubation, there were tiny colonies on MacConkey agar and blood agar, which became dry and wrinkled on further incubation. Gram stain from the colonies showed Gram-negative rods, which were oxidase-positive. Identification and antibiotic sensitivity testing were done in Automated System BD Phoenix (Becton Dickinson, USA) and disc diffusion method, which identified the organism as Burkholderia cepacia complex (95% confidence) and was sensitive to ceftazidime, chloramphenicol, co-trimoxazole, levofloxacin and meropenem. Antibiotic Sensitivity report was interpreted according to CLSI guidelines.[1] His urine and blood culture samples were sterile. Surgical debridement was done, and the patient was started on ceftazidime (1 g) and tazobactam (125 mg) intravenous every eight hourly along with oral co-trimoxazole (trimethoprim 160 mg + sulfamethoxazole 800 mg) twice daily. The patient became clinically stable and was discharged on request. However, despite surgical intervention and specific antibiotic therapy, the patient succumbed to the infection after a few days.

Case 2

First admission

A 55-year-old male was admitted to the hospital with complaints of pain and swelling (non-traumatic) of the left foot for 13 days. He was serviceman by profession and had a history of T2DM for 5 years. On examination, there was tenderness and swelling of the left ankle region. The overlying skin was oedematous and tense. He was diagnosed as a case of cellulitis with septic arthritis of the left ankle and tenosynovitis. The patient gave surgical history of undergoing transurethral resection of the prostate twice. Haematological findings (on admission) were Hb: 6.4 g/dl, TLC: 15.1 × 103/μL, ESR: 125 mm/h, glycosylated (HbA1c): 8.1% and CRP: 371.13 mg/L. USG examination showed fluid collection around the left ankle joint region, which was aspirated and sent for examination. Cytological examination of the aspirate was markedly cellular and predominantly showed polymorphonuclear inflammatory cells. He underwent abscess drainage and debridement under spinal anaesthesia. His perioperative course was uneventful. Sample processing was done as per the standard protocol, and identification and antibiotic sensitivity were done using Automated System BD Phoenix (Becton Dickinson, USA). Pus culture from abscess grew B. cepacia complex. Antibiotic sensitivity report, interpreted as per the CLSI guidelines, showed that the isolate was sensitive to ceftazidime, co-trimoxazole and meropenem.[1] His blood culture was sterile. The patient was started on ceftazidime (1 g) and tazobactam (125 mg) intravenous every 8 hourly along with oral co-trimoxazole (trimethoprim 160 mg + sulfamethoxazole 800 mg) twice daily. His general condition improved gradually, and he was discharged in a healthy condition.

Second admission

The patient was admitted again after 1 month with the complaints of intermittent fever and cough for 1 week. On examination, the patient was febrile but conscious and oriented. On examination, his HR and BP were 130/min and 100/50 mm Hg, respectively. Chest examination revealed congestion bilaterally; cardiovascular and abdominal examinations revealed no significant findings. Haematological findings were as follows: Hb: 4.9 g/dl, TLC: 300 cells/μL, ESR: 90 mm/h, CRP: 370 mg/L and procalcitonin: 27 ng/ml. Meropenem, teicoplanin and fluconazole were started along with other supportive medications. The patient's condition gradually deteriorated, and he was finally put on mechanical ventilation for respiratory insufficiency. However, the patient succumbed to the infection and died on the 2nd day of admission.

Both the isolates were sent for confirmation by molecular methods. Confirmation was done by the 16S ribosomal RNA (rRNA) study where it was identified as B. pseudomallei and was submitted to GenBank (https://www.ncbi.nlm.nih.gov/genbank/with the accession No. MH006571 and MN960591, respectively). [Figure 1] shows the phylogenetic tree constructed based on the 16S rRNA nucleotide sequences of ~1400 bp.
Figure 1: The phylogenetic tree is based on the 16S ribosomal RNA nucleotide partial sequences of the two isolates (>1350 bp size) compared to reference sequences downloaded from the NCBI database. The tree was inferred using the maximum likelihood method and Kimura 2-parameter model with a discrete gamma distribution to model evolutionary rate differences among sites after finding the best model fit. The tree is drawn to scale, with branch lengths measured in the number of substitutions per site. Evolutionary analyses were conducted in MEGA X

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 ~ Discussion Top


B. pseudomallei is a Gram-negative obligatory aerobic non-spore-forming bacillus, the aetiological agent of melioidosis, a life-threatening infection responsible for an estimated 89,000 deaths annually worldwide. This organism has been isolated from clinical samples such as blood, sputum, pus, urine, synovial, peritoneal and pericardial fluids. The most common predisposing factor for B. pseudomallei infection is diabetes mellitus followed by preexisting renal disease, thalassemia, cirrhosis and immunocompromised status. It is estimated that individuals with diabetes mellitus has over 12 times risk for melioidosis after adjustment of other risk factors.[2]

B. pseudomallei is known to be present in soil and surface waters and infection may be acquired by exposure through the broken skin, inhalation or ingestion of the organism. Both the cases reported were known diabetics presenting with musculoskeletal swelling involving the ankle joint. One case gave the history of trauma in the affected joint, which may have predisposed the infection, the other case came with swelling of the left foot with no history of recent trauma. In India, B. pseudomallei has been reported mainly from the southern states of India, namely Tamil Nadu, Kerala, Karnataka, Maharashtra, Orissa, West Bengal, Pondicherry but rarely from the North-Eastern region. Findings and outcomes of the reported cases of B. pseudomallei septic arthritis from India are shown in [Table 1].[3],[4],[5],[6],[7],[8] Pandey et al. reported that 10% relapse occurs even after 20 weeks of treatment and increases to 30% if the duration of treatment is <8 weeks.[3] The case fatality rate of melioidosis is 10%–50%.[2]
Table 1: Cases and outcome of Burkholderia pseudomallei septic arthritis cases from India

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There are reports of misidentification of B. pseudomallei as B. cepacia by automated systems.[9] Clinical and microbiological suspicion are important for the successful management of these cases. Preferred antimicrobial therapy consists of an initial intensive phase with intravenous ceftazidime or intravenous meropenem and subsequent eradication phase with oral co-trimoxazole or doxycycline. We believe that many cases of B. pseudomallei infection might have gone unrecognized or incorrectly identified. Some cases may also have been dismissed as contamination because of laboratorians' and clinicians' lack of familiarity with this bacterium. Our review suggests that advanced laboratory techniques will lead to more recognized cases and that further studies are necessary to understand this bacterium's clinical significance.

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.
Clinical and Laboratory Standards Institute. Performance Standards for Antimicrobial Susceptibility Testing. CLSI Supplement M100. 30th ed. Wayne, PA: Clinical and Laboratory Standards Institute; 2020.  Back to cited text no. 1
    
2.
Wiersinga WJ, Virk HS, Torres AG, Currie BJ, Peacock SJ, Dance DA, et al. Melioidosis. Nat Rev Dis Primers 2018;4:17107.  Back to cited text no. 2
    
3.
Pandey V, Rao SP, Rao S, Acharya KK, Chhabra SS. Burkholderia pseudomallei musculoskeletal infections (melioidosis) in India. Indian J Orthop 2010;44:216-20.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Deshmukh M, Mundhada S. Chronic suppurative joint effusion due to Burkholderia pseudomallei: A case report. Indian J Pathol Microbiol 2013;56:460-3.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Vidyalakshmi K, Shrikala B, Bharathi B, Suchitra U. Melioidosis: An under-diagnosed entity in Western Coastal India: A clinico-microbiological analysis. Indian J Med Microbiol 2007;25:245-8.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Dhodapkar R, Sujatha S, Sivasangeetha K, Prasanth G, Parija SC. Burkholderia pseudomallei infection in a patient with diabetes presenting with multiple splenic abscesses and abscess in the foot: A case report. Cases J 2008;1:224.  Back to cited text no. 6
    
7.
Rajadhyaksha A, Sonawale A, Khare S, Kalal C, Jankar R. Disseminated melioidosis presenting as septic arthritis. J Assoc Physicians India 2012;60:44-5.  Back to cited text no. 7
    
8.
Gouse M, Jayasankar V, Patole S, Veeraraghavan B, Nithyananth M. Clinical outcomes in musculoskeletal involvement of Burkholderia pseudomallei infection. Clin Orthop Surg 2017;9:386-91.  Back to cited text no. 8
    
9.
Koh TH, Yong Ng LS, Foon Ho JL, Sng LH, Wang GC, Tzer Pin Lin RV. Automated identification systems and Burkholderia pseudomallei. J Clin Microbiol 2003;41:1809.  Back to cited text no. 9
    


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