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| CASE REPORT |
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| Year : 2020 | Volume
: 17
| Issue : 1 | Page : 20-23 |
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Cervical prevertebral abscess with Burkholderia pseudomallei: A report of two cases and review of literature
Suresh Shivadasan Pillai1, Nikhil Narayanan2, Dipu Kuttippala Parambil1, Sudarsana1, Poornima Mankara Valsan1, Manikandan Thangarasu2
1 Department of spine surgery, Baby Memorial Hospital, Calicut, Kerala, India
2 Department of Orthopaedics, Baby Memorial Hospital, Calicut, Kerala, India
| Date of Submission | 31-Oct-2020 |
| Date of Decision | 10-Nov-2020 |
| Date of Acceptance | 01-Nov-2020 |
| Date of Web Publication | 17-Nov-2020 |
Correspondence Address:
Nikhil Narayanan
Chithira, Mayanad P.O, Calicut - 673 008, Kerala
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/joasis.joasis_10_20
Melioidosis is a life-threatening infectious disease caused by Burkholderia pseudomallei, seen more commonly in South East Asia and Northern Australia. Though it is an important cause of sepsis, musculoskeletal melioidosis is rare in the Indian subcontinent. We report two cases of cervical prevertebral abscess in a 60-year-old female and a 62-year-old male, caused by B. pseudomallei. No cervical prevertebral abscess due to B. pseudomallei has been reported in the literature to the best of our knowledge.
Keywords: Burkholderia pseudomallei, melioidosis, prevertebral abscess
How to cite this article:
Pillai SS, Narayanan N, Parambil DK, Sudarsana, Valsan PM, Thangarasu M. Cervical prevertebral abscess with Burkholderia pseudomallei: A report of two cases and review of literature. J Orthop Assoc South Indian States 2020;17:20-3 |
How to cite this URL:
Pillai SS, Narayanan N, Parambil DK, Sudarsana, Valsan PM, Thangarasu M. Cervical prevertebral abscess with Burkholderia pseudomallei: A report of two cases and review of literature. J Orthop Assoc South Indian States [serial online] 2020 [cited 2020 Nov 25];17:20-3. Available from: https://www.joasis.org/text.asp?2020/17/1/20/300754 |
| Introduction | |  |
Melioidosis is an infection caused by Gram-negative bacillus Burkholderia pseudomallei. The organism enters the body through ingestion, inhalation, or direct inoculation.[1] Patients usually present with abscesses (localized or multifocal) with or without sepsis. Persons with diabetes, chronic renal failure, and immunosuppression are more prone to get the disease. However, B. pseudomallei infection has been reported in immunocompetent patients also both as systemic melioidosis and localized musculoskeletal abscesses. The case fatality rates are high with melioidosis even with appropriate antibiotic treatment.
| Case Report | | |
Case 1
A 60-year-old female, with no known comorbidities, presented with neck pain and head ache of 3 weeks' duration. Her symptoms aggravated later with associated fever. Her neck movements – flexion and extension – were grossly restricted. She was admitted and further evaluated. Her initial blood investigations showed a total leukocyte count of 18,800/mm3 (4000–11,000/mm3); erythrocyte sedimentation rate of 61 mm/h (0–10 mm/h); C-reactive protein of 192 mg/dL (0–6 mg/dL); and altered liver function test revealed direct bilirubin – 0.81 mg/dL, serum glutamic pyruvic transaminase – 152 μ/l, and serum alkaline phosphatase – 233 μ/L.
Magnetic resonance imaging (MRI) cervical spine showed collection with peripheral thick rim of enhancement measuring 62 mm × 20 mm × 30 mm at transverse × anteroposterior (AP) × cranio-caudal, noted in the prevertebral space of C-2 vertebra, suggestive of abscess. Increased enhancement and T2-weighted/short-tau inversion recovery hyperintensity were noted in the retropharyngeal, prevertebral, paravertebral, and parapharyngeal spaces from C1 to C5 vertebral levels. There was loss of definition of the anterior arch of Atlas More Details, with marrow edema extending into the lateral mass [Figure 1] and [Figure 2]. | Figure 1: Magnetic resonance imaging showing prevertebral abscess of anteroposterior measurement 22 mm
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 | Figure 2: Magnetic resonance imaging showing a progression in prevertebral abscess with anteroposterior measurement of 26 mm
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She was started on intravenous (IV) antibiotics (piperacillin–tazobactam 4.5 g 8th hourly) from another center. The patient showed no signs of improvement. Hence, she was referred to our center for further management. Repeat screening MRI of cervical spine showed an increase in prevertebral abscess. The thickness progressed to 26 mm [antero-posterior thickness] at the level of c2, for which surgical drainage and culture study was planned She underwent surgical drainage of the abscess through anterior approach and the specimen was sent for microbiological study and histopathology.
The pus culture and sensitivity study of the specimen yielded moderate growth of B. pseudomallei, which was sensitive to ceftazidime, meropenem, and co-trimoxazole.
She was initiated on IV regimen of injection ceftazidime (2 g IV 8th hourly) and oral co-trimoxazole. The patient improved clinically, and laboratory parameters improved to normal limits. At follow-up, she was asymptomatic. The case was discussed with a microbiologist and an infectious disease specialist. She was advised to continue oral co-trimoxazole for 6 months in view of the notorious high recurrence rate.
Case 2
A 62-year-old male, who is a known case of Type II diabetes mellitus, presented with the complaints of upper back ache for 2 months. He was evaluated and initial blood investigations were normal except for deranged blood glucose level. MRI cervical and dorsal spine was taken which reported T2 hyperintense prevertebral collection from C2 to C5 vertebral levels, suggestive of prevertebral abscess.
[Figure 3] and [Figure 4] depict MRI cervical spine showing large multiseptated collections in the pre- and paraspinal regions from C5 to D4 and prevertebral collection from C2 to C5.
He underwent computed tomography-guided biopsy of the lesion and the specimen was sent for culture and biopsy. Fine-needle aspiration cytology of the prevertebral collection was in consistent with a granulomatous lesion. Culture and sensitivity study revealed moderate growth of B. pseudomallei, which was sensitive to ceftazidime and co-trimoxazole. He was started on IV ceftazidime (2 g 8th hourly). The patient is better with improvements in the symptoms.
| Discussion | | |
Melioidosis was first described as a glanders-like disease by a pathologist Alfred Whitmore in 1911. The term melioidosis was coined by Stanton and Fletcher in 1932. The causative organism was known by various names such as Bacillus whitmorii, Bacillus pseudomallei, Malleomyces pseudomallei, and Pseudomonas pseudomallei during the last century. It is known as B. pseudomallei since 1992. It is a slender, motile, aerobic, oxidase-positive, catalase-positive, Gram-negative bacillus with bipolar staining, found in contaminated water and soil. It will grow on most laboratory media such as sheep blood and chocolate and MacConkey agars as smooth, creamy, white colonies with a characteristic musty odor. Specific Ashdown's medium is also shown to demonstrate the growth.
Melioidosis has emerged as a major health problem in Southeast Asia and Northern Australia. The disease is being increasingly recognized in the Indian subcontinent. Persons living in endemic areas, particularly farmers in paddy fields, are more susceptible to the disease. Other common predisposing factors are diabetes, liver disease, chronic renal disease, chronic lung disease, and immunosuppression. The infection is acquired through percutaneous inoculation, inhalation, and ingestion. Clinical manifestations include pneumonia; genitourinary and soft-tissue infections; abscesses in the lung, liver, and spleen; septicemia and bacteremia without evident focus. It may mimic various diseases such as pulmonary tuberculosis and polyarthritis. Musculoskeletal melioidosis presenting as osteomyelitis and septic arthritis is rare (4%–12%). Our case was a rare presentation as cervical prevertebral abscess in an immunocompetent patient.[2] Cervical prevertebral abscess secondary to B. pseudomallei has not been reported in the literature to the best of our knowledge.
Isolation of the organism from body fluid is the gold standard for diagnosis. It is sensitive to ceftazidime, chloramphenicol, carbapenems, co-trimoxazole, and doxycycline, but resistant to macrolides, aminoglycosides, fluoroquinolones, and second-generation cephalosporins. Previously, ceftazidime was considered the single best drug for melioidosis. Now, the standard treatment regimen is intensive therapy with at least 2 weeks of IV meropenem or ceftazidime, followed by oral co-trimoxazole and doxycycline (eradication regimen) for 3–6 months. Recurrence rate after treatment is around 1 in 16 patients and mortality rates are as high as 14%–40%.
Prompt identification of organism and institution of appropriate antibiotic regimen helps to teduce the mortality of melioidosis.
Our first patient hails from a village, and she lives in a house near a paddy field and used to work there. She had persistent neck pain and difficulty in swallowing. Melioidosis is a differential diagnosis in patients with similar history and prevertebral abscess. This can easily be mistaken for tuberculosis by histology for the unwary.
The second patient did not have a history of living near a paddy field nor working in a paddy field. He is a type II diabetes mellitus patient with very poor diabetic control, making him prone to immunocompromised.
It is advisable and mandatory to get tissue diagnosis and culture and sensitivity reports before embarking on empirical antibiotic therapy.[3] Melioidosis is a chronic infection that requires long-term antibiotic treatment and follow-up.[Figure 3] and [Figure 4]
[Figure 5] is bipolar staining and safety pin appearance of organism. [Figure 6] is mri c spine of case 2 patient showing pre vertebral abscess. | Figure 5: Bipolar staining and safety pin appearance of Burkholderia pseudomallei  n light microscopy 100x zoom
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 | Figure 6: MRI cervical spine of case 2 patient showing pre vertebral abscess
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| Conclusion | | |
B. pseudomallei causes chronic musculoskeletal and soft-tissue infections.
Prevertebral cervical abscess without infection of spine has not been reported in the literature to the best of our knowledge.[4]
Melioidosis is known to cause recurrence if improperly treated. They have a predilection in patients with diabetes. However, it can also be caused in immunocompetent patients, like our first case. History of work in a paddy field is an important clue here.[5]
Biopsy and culture of all abscesses are mandatory and advisable before putting the patient on an empirical antibiotic therapy. If left untreated, patients with melioidosis are prone to succumb to the disease. The case fatality rate is about 10%–50% (Birnie et al., The LancetInfectious Diseases, July, 2019). Inadequately treated patients with melioidosis can have recurrence, which is often fatal.[6]
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 | | |
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| 2. | Lee HS, Riyaaz AA, Yeoh SH. Acute disseminated melioidosis presenting with septic arthritis and diffuse pulmonary consolidation in an otherwise healthy adult-a case report. Int J Med Students 2014;3:59-62. |
| 3. | Redondo MC, Gomez M, Landaeta ME. Melioidosis presenting as sepsis syndrome: a case report. International Journal of Infectious Diseases 2011;15:e217-8. |
| 4. | Rodrigo K. Melioidosis as a cause of femoral osteomyelitis and multifocal intramuscular abscess around the hip joint in a farmer: A case report. Sri Lankan J Infect Dis 2013;3:50-4. |
| 5. | Koshy M. Melioidosis: An emerging infection in India. Curr Med Issues 2020;18:94-7. [Full text] |
| 6. | Birnie E, Virk HS, Savelkoel J, Spijker R, Bertherat E, Dance DAB, et al. Global burden of melioidosis in 2015: a systematic review and data synthesis. Lancet Infect Dis 2019;19:892-902. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]
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