Translate this page into:
Disseminated Burkholderia pseudomallei infection with isolated humeral osteomyelitis - A case report
, Rakesh T. Parakadavathua, Anita Vincentb, Akhil K. Thomasa, P. Saranga
∗Corresponding author: C.R. Jithin. drjithincradhakrishnan@gmail.com
-
Received: ,
Accepted: ,
This article was originally published by Reed Elsevier India Pvt. Ltd. and was migrated to Scientific Scholar after the change of Publisher.
Abstract
Abstract
In the Indian subcontinent, Burkholderia pseudomallei is a relatively new pathogen. Due to its ability to mimic the microorganisms that cause osteomyelitis, septic arthritis, cold abscess, pneumonia, and septicemia, it poses a dilemma for the treating surgeon on making correct diagnosis and should be considered as a differential diagnosis in endemic areas.
65-year-old male, diabetic, from north Malabar, Kerala state who presented with one month history of pain and swelling over the left arm, hoarseness of voice and mild dysphagia to liquids. Burkholderia pseudomallei was in pus collected from abscess and underlying osteomyelitis in left arm. He recovered completely after getting treatment with prolonged course of intravenous ceftazidime and later oral co-trimoxazole.
The major hurdle in treating Melioidosis remains the delay in making the right diagnosis and providing adequate treatment. As it can be easily mistaken for tuberculosis, high degree of suspicion is necessary, as early detection and management of the disease can lead to better outcomes.
Keywords
Burkholderiapseudomallei
Melioidosis
Safety pin appearance
Osteomyelitis
1 Introduction
Melioidosis, or Whitmore's disease, is caused by a ubiquitous gram negative soil pathogen, Burkholderia pseudomallei. The clinical spectrum ranging from pneumonia, cutaneous infection to disseminated disease with fulminant septicemia.1 As it is a close mimicker of tuberculosis and other gram negative infections, considerable diagnostic dilemma occurs before correct diagnosis is established. Incidence is high in Australia and tropical Southeast Asian nations compared to rest of world. In India, it is increasingly reported from the coastal regions of TamilNadu and Kerala, two southern states with tropical climate.2 Rare case reports from other parts of the world indicate that infection could be present with an under estimated incidence.3
2 Case report
A 65-year-old male known case of type 2 diabetes presented with insidious onset of pain and swelling over the left arm for one month. He later developed hoarseness of voice, coughing during liquid intake progressing to difficulty to swallowing of liquids. He also gave a history of right sided cervical lymphadenopathy 2 months before presentation, which was reported as suppurative lymphadenitis after an aspiration (FNAC). On examination, his vitals were stable, afebrile, with a prominent soft nontender right cervical lymph node of 3 × 4 cm. Systemic examination was normal except for hoarseness of voice. On local examination, there was a firm swelling over the middle third of the left arm with tenderness. X-ray showed hypodense area at the junction of middle and distal third junction of left humerus (Fig. 1). The plain CT left humerus was suggestive of acute osteomyelitis with abscess formation. Screening MRI showed extensive marrow edema involving the middle and distal third of the left humerus with a large ill-defined periosteal collection involving the adjacent brachialis and brachioradialis muscle with extensive surrounding soft tissue edema (Fig. 2). CT neck and thorax showed left vocal cord palsy, suppurative lymphadenitis involving right level IB and II of the neck with reactive changes in adjacent lung. The patient was admitted for debridement and curettage of the left humerus. Thick caseous pus was drained, and the sample taken during curettage was later sent for investigations (Fig. 3). Biopsy from left humerus curettage showed fragments of fibro collagenous tissue, granulation tissue with areas of necrosis, collections of foamy histiocytes, and a few loose aggregates of epithelioid histiocytes, neutrophils, and multi-nucleated giant cells. It was reported as pyogranulomatous lesions (Fig. 4) rising the possibility of tuberculosis infection, though AFB staining and PCR for Mycobacterium tuberculosis were negative. Gram staining of the pus aspirate showed many pus cells, a few gram-positive cocci in pairs and chains, and a few gram-negative bacilli with a safety pin appearance. The gram-negative growth was identified as Burkholderia pseudomallei (Fig. 5). The patient was diagnosed with disseminated Burkholderia pseudomallei infection involving left humerus, left lung,para-aortic,cervical lymph nodes causing right recurrent laryngeal nerve palsy. The patient was started on Inj. Ceftazidime 2g IV 8th hourly 6 weeks followed by oral cotrimoxazole for a minimum of nine months with strict diabetes control. Oral cotrimoxazole was added on the fourth week of therapy as we thought some sluggishness in response to therapy. In eight weeks neck lesions healed, and the humerus showed no more pus discharge. The patient was discharged with advice to continue oral cotrimoxazole 960 mg tablet twice daily for total period of 9 months. There were no radiological features on x ray suggestive of osteomyelitis focus on 6th (Fig. 6) and on 12th month follow up. Cervical lymphadenopathy completely resolved with full recovery of recurrent laryngeal nerve palsy without any variation in vocal quality and the surgical scar appears healthy (Fig. 7).
3 Discussion
Melioidosis occurs due to infection of Burkholderia pseudomallei, a gram-negative aerobic, motile bacteria. Pathologist Alfred Whitmore initially identified Melioidosis in a case series of 38 patients in Rangoon, Burma (currently Myanmar), in 1912.4 Musculoskeletal melioidosis closely mimics other infections both clinically and radiologically and diagnosis requires a high index of clinical suspicion and confirmed on proving the microbiology.5 In eastern Asia and northern Australia, it is a significant contributor to sepsis. Melioidosis,a more indolent form characterized by the development of abscesses, particularly in the prostate, skeletal muscle, liver, and lungs.6 Thalassemia, diabetes mellitus, pre-existing renal conditions, and occupational exposure remain as significant risk factors for Melioidosis. Compared to nonbacteremic, the only significant predictor linked with bacteremic Melioidosis was diabetes mellitus.7 Melioidosis has the potential to spread to new and productive pastures as the global movement by humans and other animals continue to increase.8 To lessen the impact of this illness, studies examining the role of prevention strategies, early clinical detection, and better management of severe sepsis are needed.3 A 65-year-old male known case of type 2 diabetes from north Malabar, Kerala sate presented with pain and swelling over the left arm with hoarseness of voice and mild dysphagia to liquids for a month duration, disseminated Burkholderia pseudomallei infection was identified and recovered completely after treatment with intravenous ceftazidime and oral co-trimoxazole. Standard line of treatment for Melioidosis include IV therapy with ceftazidime for 2–4 weeks followed by oral trimethoprim-sulfamethoxazole (TMP-SMX) for 12–20 weeks. All-cause mortality was shown to be considerably lower in the 12-week regimen group than in the 20-week regimen group [0.3%] vs [3%], respectively.9
4 Conclusion
Burkholderia pseudomallei continues to pose diagnostic challenge in endemic countries. This can only be improved with better awareness of the disease and early consideration into the of differential diagnoses. As it can be easily mistaken for tuberculosis, early diagnosis remain the key step in managing this completely treatable illness.
Funding/sponsorship
This research did not receive any specific grant from funding agencies in the public, commercial or not-for-profit sectors.
Informed consent (patient/guardian)
Detailed written informed consent obtained for publication of data, images, and treatment-related documents without any objection.
Authors' contribution
All authors equally contributed to Conceptualization, Data curation, Format analysis, Investigation, Methodology, Software, Supervision, Validation, Visualization, Writing – review & editing.
Ethical approval
Not applicable.
Availability of data and materials
All datas including images are available whenever requested.
References
- A rare case of a knee septic arthritis by Burkholderia pseudomallei: a case report from a Tertiary Care Hospital of Andaman and Nicobar Island. J Orthop Case Rep. 2021 Sep;11(9):20-23.
- [Google Scholar]
- Melioidosis: epidemiology, pathophysiology, and management. Clin Microbiol Rev. 2005;18(2):383-416.
- [Google Scholar]
- Melioidosis: a review of orthopedic manifestations, clinical features, diagnosis and management. Indian J Med Sci. 2007 Oct;61(10):580-590.
- [Google Scholar]
- Risk factors for melioidosis and bacteremic melioidosis. Clin Infect Dis. 1999;29(2):408-413.
- [Google Scholar]
- A comparison between 12 versus 20 Weeks of trimethoprim-sulfamethoxazole as oral eradication treatment for melioidosis: an open-label, pragmatic, multicenter, non-inferiority, randomized controlled trial. Clin Infect Dis. 2021;73(11):e3627-e3633.
- [Google Scholar]