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Table of Contents
CASE REPORT
Year : 2021  |  Volume : 10  |  Issue : 1  |  Page : 43-46

Chronic osteomyelitis of the femur caused by Burkholderia pseudomallei in a patient with type 2 diabetes mellitus


1 Department of Orthopaedics, Nizam's Institute of Medical Sciences, Hyderabad, Telangana, India
2 Department of General Medicine, Nizam's Institute of Medical Sciences, Hyderabad, Telangana, India

Date of Submission14-Oct-2019
Date of Decision04-May-2020
Date of Acceptance17-Jul-2020
Date of Web Publication4-Mar-2021

Correspondence Address:
Maddali Dheerj Manikanta
Junior Consultant in Department of Orthopedics, Nizams Institute of Medical Sciences, Hyderabad, Telangana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JCSR.JCSR_112_19

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  Abstract 


Melidiosis is a life-threatening infectious disease, and the causative organism is Burkholderia pseudomallei. It occurs as an environmental saprophyte in endemic regions of northern Australia and Southeast Asia. A 43-year-old male patient known to have type 2 diabetes mellitus, presented with fever, thigh swelling. He had received treatment with multiple antimicrobial agents, including antituberculosis drugs, but no improvement was observed. An incision and drainage of medullary canal was done and pus culture grew Burkholderia pseudomallei. He was managed with intravenous meropenem followed by oral cotrimoxazole. A high index of suspicion is required for early diagnosis. Burkholderia pseudomallei infection should be suspected in patients with diabetes mellitus, in patients presenting with an abscess not responding to antimicrobial treatment, especially in patients from endemic areas. The organism responds only to specific antibiotics; therefore correct and timely diagnosis becomes crucial for a better outcome.

Keywords: Burkholderia pseudomallei, melioidosis, osteomyelitis


How to cite this article:
Iyengar R, Manikanta MD, Goveen M. Chronic osteomyelitis of the femur caused by Burkholderia pseudomallei in a patient with type 2 diabetes mellitus. J Clin Sci Res 2021;10:43-6

How to cite this URL:
Iyengar R, Manikanta MD, Goveen M. Chronic osteomyelitis of the femur caused by Burkholderia pseudomallei in a patient with type 2 diabetes mellitus. J Clin Sci Res [serial online] 2021 [cited 2021 Aug 3];10:43-6. Available from: https://www.jcsr.co.in/text.asp?2021/10/1/43/310762




  Introduction Top


Burkholderia pseudomallei is the causative agent of emerging infectious disease, melioidosis, which is endemic in the tropical regions of the world.[1],[2],[3] The majority of patients with identified risk factors such as diabetes mellitus, heavy alcohol use, malignancy, chronic lung and kidney disease, corticosteroid use, thalassemia, rheumatic heart disease, systemic lupus erythematosus and cardiac failure acquire this organism through percutaneous inoculation or inhalation. The clinical manifestations are variable, ranging from localised abscess formation to septicaemia. Melioidotic bone and joint infections are rarely reported, but are an established entity. Here, we report a case of chronic osteomyelitis of the femur in a patient with type 2 diabetes mellitus who presented with a non-healing abscess.


  Case Report Top


A 43-year-old male, known to have type 2 diabetes mellitus, presented to Nizam's Institute of Medical Sciences with complaints of pain in the right hip and thigh and difficulty in walking for the last 3 months. He gave a history of fever, weight loss of 15 kg over the previous 3 months and loss of appetite. On examination, he was febrile; vitals parameters normal; cardiovascular, abdominal, respiratory and central nervous systems were unremarkable.

On local examination, diffuse swelling of the right thigh extending from the gluteal region to the mid-thigh region was evident; the swelling was warm and tender. There was no inguinal lymphadenopathy. The range of movements of the right hip joint was restricted and painful. The skin over the thigh region was normal; there were no scars and sinuses.

On laboratory evaluation, haemoglobin was 9.7 g/dL, total leucocyte count was 11,300/mm3, fasting and postprandial blood glucose levels were 156 mg/dL and 315 mg/dl respectively; glycosylated haemoglobin was 9.3%. Erythrocyte sedimentation rate was 150 mm at the end of first hour. Serum ferritin was 1790 mg/dL, liver function tests and renal function tests were normal and he was human immunodeficiency virus seronegative.

Radiograph of the right hip with thigh showed areas of radiolucency involving the neck and greater trochanter of the femur, periosteal reaction and sequestrum in the middle third shaft femur. Magnetic resonance imaging (MRI) of the right thigh showed findings of T2 heterogeneous hyperintensity noted involving the entire right femur suggestive of marrow oedema changes, multiple fluid pockets in vastus medialis and lateralis suggestive of an intramuscular abscess [Figure 1]. Skeletal scintigraphy showed a diffuse but nonhomogeneous moderate hotspot along the entire right femur, including head and neck [Figure 2]. Incision and drainage of the abscess and sequestrectomy of the proximal femur were done. The surgery was planned with an exploration of medullary canal and debriding the medullary canal by reaming and cleaning repeatedly with hydrogen peroxide. Sequestrectomy was contemplated with a window in the lateral cortex of the femur and exteriorisation of the sequestrum. The pus and the material were sent to microbiology, where the culture showed Gram-negative bacterium, B. pseudomallei [Figure 3]; the patient was started on intravenous (iv) ceftazidime 2 g every 6th hourly, for 14 days. Fever subsided, and the patient was discharged on oral cotrimoxazole. The patient developed pain and swelling in the distal thigh after 15 days for which iv meropenem 2 g every 8th hourly was started, following which the swelling and pain subsided. Cotrimoxazole double-strength tablets were continued for 6 months. He underwent MRI after 10 months and was found to have a residual infection [Figure 4]. Hence, oral cotrimoxazole was given for a further period of one year.
Figure 1: Magnetic resonance imaging T2/STIR showing heterogeneous hyperintensity noted involving the entire right femur suggestive of marrow oedema changes, multiple fluid pockets in vastus medialis and lateralis suggestive of an intramuscular abscess STIR = Short-Tau inversion recovery

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Figure 2: Magnetic resonance imaging T2/STIR image showing decrease in hyperintensity of marrow after incision and drainage of medullary canal of the femur but with residual infection STIR = Short-Tau inversion recovery

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Figure 3: Ashdown media showing growth of purple wrinkled colonies of Burkholderia pseudomallei

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Figure 4: Skeletal scintigraphy showing diffuse but nonhomogeneous hotspot involving the entire right femur including head and neck

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  Discussion Top


B. pseudomallei is the causative agent of a severe and fatal infectious disease, which is called melioidosis. B. pseudomallei, a Gram-negative bacterium, is a water and soil pathogen in Eastern Asia and Northern Australia.[4],[5],[6] Melioidosis has emerged as an important cause of morbidity and mortality in the Southeast. B. pseudomallei can infect healthy individuals, but it is seen more commonly in patients with comorbidities such as malignancy, immunosuppression and diabetes mellitus, as in our case. Clinical manifestations of melioidosis range from subclinical infection to localised cutaneous lesions, subacute pneumonia, bone and joint infections, abscesses in body organs and cranial abscesses to life-threatening septicaemia.[4]

The clinical presentations of bone and joint infections due to B. pseudomallei are indistinguishable from other aetiologies[7],[8] of osteomyelitis. Melioidosis should be considered in patients from the endemic disease area or who are returning from these areas.[9] Early diagnosis can be made by sending pus for culture to microbiological laboratory and start prompt administration of appropriate antimicrobial therapy.[10],[11],[12],[13],[14] Mortality in acute severe melioidosis, even with proper treatment, remains considerably high, ranging from 30% to 47%. Melioidotic bone and joint infections, though an established entity, are rarely reported.[15],[16] The knee joint is the most commonly involved joint in melioidosis, followed by the ankle, hip and shoulder joints. Many cases have been linked to occupational and recreational exposure to surface water. Melioidosis is diagnosed on the basis of the clinical and laboratory parameters and radiology. A majority of patients have increased or low white blood cell and neutrophil cell counts, increased C-reactive protein, serum procalcitonin, erythrocyte sedimentation rate, impaired renal or liver functions, and in some cases, organ failure Accurate and timely diagnosis remains crucial to initiate prompt definitive antimicrobial therapy to reduce the mortality. Isolation of B. pseudomallei from the clinical specimens is considered to be the gold standard in the diagnosis of melioidosis.[13]

Treatment is vigorous and repeated washouts, and several extensive debridements of infected bone are the cornerstones of the treatment of bone and joint infections. Prompt administration of antibiotics remains essential to reduce morbidity and mortality. The blood sugars are controlled by insulin injection preoperatively. Intravenous antimicrobial therapy should be prolonged for deep-seated infection or complicated infections from four to 8 weeks, followed by oral maintenance therapy for a minimum of 12 weeks. Ceftazidime (50 mg/kg/day maximum of 2 g), or a member of the carbapenem group, is the drug of choice to treat this infection, followed by oral cotrimoxazole as a monotherapy or amoxicillin–clavulanic acid. Use of ceftazidime, meropenem or imipenem and trimethoprim–sulfamethoxazole combination therapy with folic acid in the acute phase followed by any two antibiotics from the group of trimethoprim–sulfamethoxazole combination therapy, doxycycline, and amoxicillin–clavulanic acid, as eradication therapy is indicated in deep-seated infections. The patient was changed over to o oral metformin after the surgery and blood glucose level were under control.

In endemic areas, B. pseudomallei should be considered as one of the causative agents of bone and joint infections because of its rising incidence and high rate of morbidity and mortality if not diagnosed and treated early on.[12] A high index of suspicion of melioidosis is required to make the diagnosis. Isolation of B. pseudomallei from clinical specimens considered to be the gold standard; however, to achieve this, samples must be processed carefully and in the appropriate media. Treatment of melioidosis affecting bones and joints consists of antimicrobial therapy coupled with surgical management including washouts of joints and debridement of infected bone.[13] Those patients with deep-seated or complicated infections require intravenous antibiotics for 4–8 weeks, followed by oral antibiotics for a minimum of 12 weeks.[14] Ceftazidime is usually the intravenous antibiotic of choice, which is followed by oral therapy such as cotrimoxazole.[17] Unfortunately, no vaccine has yet been developed for this disease, which makes the awareness and understanding of melioidotic bone and joint infections, and the need for timely diagnosis and treatment. In bones, femur and tibia are common.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Punyagupta S. Melioidosis: A great imitator. Ramathibodi Med J 1983;6:147-53.  Back to cited text no. 1
    
2.
Whitmore A, Krishnaswami CS. An account of the discovery of a hithertoun described infective disease occurring among the population of Rangoon. Indian Med Gaz 1912;47:262-7.  Back to cited text no. 2
    
3.
Leelarasamee A, Bovornkitti S. Melioidosis: Review and update. Rev Infect Dis 1989;11:413-25.  Back to cited text no. 3
    
4.
Chaowagul W, White NJ, Dance DA, Wattanagoon Y, Naigowit P, Davis TM, et al. Melioidosis: A major cause of community-acquired septicemia in Northeastern Thailand. J Infect Dis 1989;159:890-9.  Back to cited text no. 4
    
5.
Currie BJ, Fisher DA, Howard DM, Burrow JN, Lo D, Selva-Nayagam S, et al. Endemic melioidosis in tropical northern Australia: A 10-year prospective study and review of the literature. Clin Infect Dis 2000;31:981-6.  Back to cited text no. 5
    
6.
Rode JW, Webling DD. Melioidosis in the Northern Territory of Australia. Med J Aust 1981;1:181-4.  Back to cited text no. 6
    
7.
Mukhopadhyay C, Dey A, Sugandhi Rao P, Pandey V, Sripathi Rao P. Aetiology and management of chronic granulomatous osteomyelitis: look before you leap. Singapore Med J 2007;48:e40-2.  Back to cited text no. 7
    
8.
Jayanetra P, Pipatanagul S, Punyagupta S, Ratanabanangkoon K, Varavithya W. Pseudomonas pseudomallei: 1. Infection in Thailand. Southeast Asian J Trop Med Public Health 1974;5:487-91.  Back to cited text no. 8
    
9.
Thin RN, Brown M, Stewart JB, Garrett CJ. Melioidosis: A report of ten cases. Q J Med 1970;39:115-27.  Back to cited text no. 9
    
10.
Leelarasamee A. Epidemiology of melioidosis. J Infect Dis Antimicrob Agents 1986;3:84-93.  Back to cited text no. 10
    
11.
McCormick JB, Weaver RE, Hayes PS, Boyce JM, Feldman RA. Wound infection by an indigenous Pseudomonas pseudomallei-like organism isolated from the soil: Case report and epidemiologic study. J Infect Dis 1977;135:103-7.  Back to cited text no. 11
    
12.
Jesudason MV, Shanthakumari R, John TJ. Burkholderia pseudomallei an emerging pathogen in India. Indian J Med Microbiol 1997;15:1-2.  Back to cited text no. 12
    
13.
Piggott JA, Hochholzer L. Human melioidosis. A histopathologic study of acute and chronic melioidosis. Arch Pathol 1970;90:101-11.  Back to cited text no. 13
    
14.
Ashdown LR. Relationship and significance of specific immunoglobulin M antibody response in clinical and subclinical melioidosis. J Clin Microbiol 1981;14:361-4.  Back to cited text no. 14
    
15.
Rao PS, Dhawan R, Shivananda PG. Burkholderia pseudomallei infections: Case reports of five cases. Trop Doct 2002;32:174-5.  Back to cited text no. 15
    
16.
Kanungo R, Padhan P, Bhattacharya S, Srimannarayanan J, Jayanthi S, Swaminath RP. Melioidosis: A case report from Pondicherry, South India. J Assoc Physicians India 2002;50:1438-9.  Back to cited text no. 16
    
17.
White NJ, Dance DA, Chaowagul W, Wattanagoon Y, Wuthiekanun V, Pitakwatchara N. Halving of mortality of severe melioidosis by ceftazidime. Lancet 1989;2:697-701.  Back to cited text no. 17
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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