Unusual case of surgical site infection with uncommon organism – Burkholderia pseudomallei

Fawaz Mohammed Manu; Anwar Marthya; Shameez Muhammed Salim; Vinu Elias

doi:10.4103/joasis.joasis_27_21

CASE SERIES

Year : 2022 | Volume : 19 | Issue : 1 | Page : 44-47

Unusual case of surgical site infection with uncommon organism – Burkholderia pseudomallei

Fawaz Mohammed Manu, Anwar Marthya, Shameez Muhammed Salim, Vinu Elias
Department of Orthopaedics, Iqraa International Hospital and Research Centre; Diplomate of National Board, National Board of Examination (University), Kozhikode, Kerala, India

Date of Submission	19-Nov-2021
Date of Acceptance	04-Dec-2021
Date of Web Publication	26-Jun-2022

Correspondence Address:
Shameez Muhammed Salim
Department of Orthopedics, Iqraa International Hospital and Research Centre, Malaparamba Calicut, 673009, Kerala
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/joasis.joasis_27_21

Abstract

Melioidosis is an infection caused by a facultative intracellular Gram-negative bacterium, Burkholderia pseudomallei, previously termed as Pseudomonas pseudomallei. This case report is an unusual case of surgical site infection with uncommon organism-B. pseudomallei. We report a case of melioidosis. A 62-year-old patient complained of swelling, redness, discharge from the surgical site right hip, and on and off generalized temperature from the last 15 days. The patient was toxic. Erythrocyte sedimentation rate was 130, C-reactive protein – 92, and Hb – 9.2, and on further blood investigation, the patient was diagnosed with surgical site infection with an implant in situ and underwent implant removal and surgical debridement of right hip, by intraoperative pus culture shows Staphylococcus aureus growth, and the patient was started on intravenous cefoperazone-sulbactam 1.5 g for 1 week, following which symptoms reappear. Later, Gram-stain shows the growth of B. pseudomallei. He was started on injection ceftazidime 2 g thrice daily (TDS) (Q8 hourly) for 2 weeks followed by oral cotrimoxazole for the next 9 months, the patient is on regular follow-up. Diagnosis of melioidosis is missed in many parts of the world due to lack of awareness of this infection caused by B. pseudomallei. Delay in diagnosis or treatment against melioidosis can worsen the outcome. With increasing awareness and better diagnostic facilities, probably musculoskeletal melioidosis will be increasingly diagnosed in future.

Keywords: Burkholderia pseudomallei, melioidosis, septicemia

How to cite this article:
Manu FM, Marthya A, Salim SM, Elias V. Unusual case of surgical site infection with uncommon organism – Burkholderia pseudomallei. J Orthop Assoc South Indian States 2022;19:44-7

How to cite this URL:
Manu FM, Marthya A, Salim SM, Elias V. Unusual case of surgical site infection with uncommon organism – Burkholderia pseudomallei. J Orthop Assoc South Indian States [serial online] 2022 [cited 2023 Mar 27];19:44-7. Available from: https://www.joasis.org/text.asp?2022/19/1/44/348314

Introduction

Melioidosis is an infection caused by a facultative intracellular Gram-negative bacterium, Burkholderia pseudomallei,^[1] previously termed Pseudomonas pseudomallei. It can be presented as septicemia, localized infection with/without septicemia, asymptomatic infections, ulcers, pneumonia, visceral abscesses, neurological infection, and musculoskeletal infections and can involve any organ.

It was first diagnosed by Whitmore and Krishnaswami in 1911 in Burma.^[2] It is a soil saprophyte, present in stagnant water and paddy fields, and infection is through the skin through abrasions or inhalation.^[3] Patients with diabetes mellitus, chronic renal failure, alcoholism, cirrhosis, and immunocompromised status are more susceptible^[4],[5] high-risk patient for suspected melioidosis. The most common risk factor predisposing individuals to melioidosis is diabetes mellitus, which is present in >50% of all patients with melioidosis worldwide. Individuals with diabetes mellitus have a 100-fold higher risk of melioidosis after adjustment for age, sex, and other risk factors. Other known risk factors include exposure to soil or water (especially during the rainy season, abundant in soil at depths of 10 cm from the surface), male sex (probably because of a greater risk of environmental exposure), age of >45 years, excess alcohol consumption and liver disease, chronic lung disease, chronic kidney disease, and thalassemia (which probably causes neutrophil dysfunction due to iron overload). Prolonged steroid use and immunosuppression can also predispose individuals to infection. Nonetheless, >80% of pediatric patients and ~20% of adult patients have no recognized risk factors. Melioidosis in adults who have no risk factors generally occurs in those who have been exposed to a high bacterial load, for example, by aspiration of surface water.

Clinical manifestations of melioidosis range from latent infection, localized cutaneous lesions, subacute pneumonia, bone and joint infections, abscesses in body organs, and cranial abscesses to life-threatening septicemia.^[6],[7]

B. pseudomallei is not fastidious and grows on a large variety of culture media (blood agar, MacConkey agar, eosin methylene blue, etc.). Ashdown's medium (or Burkholderia cepacia medium) may be used for selective isolation. Cultures typically become positive in 24–48 h (this rapid growth rate differentiates the organism from Burkholderia mallei, which typically takes a minimum of 72 h to grow). Colonies are wrinkled, have a metallic appearance, and possess an earthy odor. On Gram staining, the organism is a Gram-negative rod with a characteristic “safety pin” appearance (bipolar staining).^[8] On sensitivity testing, the organism appears highly resistant (it is innately resistant to a large number of antibiotics including colistin and gentamicin) and that again differentiates it from B. mallei, which is in contrast, exquisitely sensitive to a large number of antibiotics. For environmental specimens only, differentiation from the nonpathogenic Burkholderia thailandensis using an arabinose test is necessary (B. thailandensis is never isolated from clinical specimens).

The large, wrinkled colonies look like environmental contaminants, so they are often discarded as being of no clinical significance. The organism grows more slowly than other bacteria that may be present in clinical specimens and those from nonsterile sites. Nonsterile specimens should, therefore, be cultured on selective media (e.g., Ashdown's or B. cepacia medium). For heavily contaminated samples, such as feces, a modified version of Ashdown's that includes norfloxacin, amoxicillin, and polymyxin B has been proposed. In blood culture, the BacT/ALERT MB system (normally used for culturing mycobacteria) by BioMERT MB has been shown to have superior yields compared to conventional blood culture media.

Routine biochemical methods for identification of bacteria vary widely in their identification of this organism: the API ZONE system accurately identifies B. pseudomallei in 99% of cases, as does the automated VITEK 1 system, but the automated VITEK 2 system only identifies 19% of isolates. The pattern of resistance to antimicrobials is distinctive and helps to differentiate the organism from Pseudomonas aeruginosa. The majority of B. pseudomallei isolates are intrinsically resistant to all aminoglycosides (through an efflux pump mechanism) but sensitive to co-amoxiclav: this pattern of resistance almost never occurs in P. aeruginosa and is helpful in identification. Molecular methods (polymerase chain reaction) of diagnosis are possible but not routinely available for clinical diagnosis.^[9] Fluorescence in situ hybridization has also been described but has not been clinically validated, and it is not commercially available. Musculoskeletal infection due to melioidosis is not common in India. However, several cases of soft-tissue infection have been reported in the past.^{[10],[11],[12]}

Clinically, it mimics pyogenic bacterial infection, Gram-negative sepsis, tuberculosis, or even polyarthritis.^{[13],[14],[15]} An abscess may heal after incision and drainage but may recur. The patient may present with florid pneumonia which rapidly progresses to fulminant septicemia with abscess, osteomyelitis, or septic arthritis. The diagnosis is likely to be missed by the clinician and microbiologist unless a high degree of suspicion is maintained. Histopathology may show necrotizing granuloma without acid-fast bacilli, which may confuse the picture with tuberculosis.^[16] It is sensitive to ceftazidime, amoxy-clavulanic acid, cotrimoxazole, and doxycycline and resistant to aminoglycosides, macrolides, second-generation cephalosporins, fluoroquinolones, and rifamycins. Serology may be helpful in cases of culture-negative results, or in the absence of clinical samples from patients with melioidosis. However, the serology results should be interpreted cautiously in endemic areas, where local populations have raised melioidosis antibody levels.

Case Report

A 62-year-old patient, businessman by occupation, hailing from Vadakara, Kerala, with type 2 DM for more than 10 years with poor glycemic control, presented with a history of fall in household 6 months back and sustained injury to right hip and right elbow, he was diagnosed to have right intertrochanteric fracture and comminuted olecranon fracture, for which he underwent closed reduction internal fixation with proximal femoral nailing and tension band wiring in olecranon.

Now, from the last 1 month, he complained of swelling, redness, discharge from the surgical site right hip, and on and off generalized temperature from the last 15 days [Figure 1].

Figure 1: Surgical site right hip

Click here to view

Clinically, the patient was toxic (shows features of infection).

Erythrocyte sedimentation rate was 130, C-reactive protein – 92, and Hb – 9.2, and on further blood investigation, the patient was diagnosed with surgical site infection with implant in situ and underwent implant removal and surgical debridement of the right hip, by intraoperative pus culture shows Staphylococcus aureus growth, and the patient was started on intravenous cefoperazone–sulbactam 1.5 g for 1 week, following which symptoms reappear.

Later, Gram-stain shows the growth of B. pseudomallei. Serial blood cultures grew Gram-negative bacilli, later identified as B. pseudomallei, and diagnosed to have melioidosis [Figure 2], following which he was started on injection ceftazidime 2 g TDS (Q8 hourly) for 2 weeks followed by oral cotrimoxazole for the next 9 months, the patient is on regular follow-up.

Figure 2: Gram-stain shows growth of B.pseudomallei.

Click here to view

Discussion

Melioidosis is caused by B. pseudomallei, which is a facultative intracellular Gram-negative, saprophytic bacterium, commonly found in soil or contaminated water.

The populations at risk are those who have occupational exposure to wet soil or surface water in the form of farming, agriculture, gardening, fishing, manual labor, land surveying, building construction, and immunocompromised states such as diabetes, alcoholism, chronic renal failure, chronic lung disease, and HIV/AIDS.^{[17],[18],[19],[20],[21]}

Direct inoculation (especially through breaks in skin) is known to be the major mode of transmission of infection. Inhalation, ingestion, and person-to-person transmission are other common modes of transmission, and sexual transmission and vertical transmission at childbirth have also been reported.^{[22],[23],[24],[25],[26]} It has a significant mortality rate despite treatment, and also it is known to cause reinfection and recurrences.

Melioidosis can have two major presentations: acute infection (symptoms lasting <2 months) and chronic infections (symptoms lasting more than 2 months). Melioidosis can present as septicemia, localized infection with/without septicemia, asymptomatic infections, ulcers, pneumonia, visceral abscesses, neurologic infection, and musculoskeletal infections.

Musculoskeletal melioidosis is a well-recognized manifestation of the disease.^[7],[12] It can manifest as soft-tissue abscesses, septic arthritis, spondylitis, sacroiliitis, and osteomyelitis.

Conclusion

Diagnosis of melioidosis is missed in many parts of the world due to a lack of awareness of this infection caused by B. pseudomallei. Delay in diagnosis or treatment against melioidosis can worsen the outcome. Initial therapy with intravenous antibiotics followed by oral maintenance therapy and appropriate surgical intervention remains vital in the management. 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. Ceftazidime is usually the intravenous antibiotic of choice, which is followed by oral therapy such as cotrimoxazole. As no vaccine is yet available for melioidosis, the early dignosis of melioidotic bone and joint innfection and prompt management is of atmost importance. With increasing awareness and better diagnostic facilities, probably musculoskeletal melioidosis will be increasingly diagnosed in future.

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.

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