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Table of Contents    
CASE REPORT
Year : 2022  |  Volume : 70  |  Issue : 2  |  Page : 778-780

CNS Melioidosis: A Diagnostic Challenge


1 Department- Critical Care and Neurology, Institute of Neurosciences Kolkata, Kolkata, West Bengal, India
2 Department of Neurology, Institute of Neurosciences Kolkata, 185/1 A.J.C. Bose Road, Kolkata, West Bengal, India

Date of Submission13-Oct-2020
Date of Decision13-Feb-2021
Date of Acceptance15-May-2021
Date of Web Publication3-May-2022

Correspondence Address:
Dr. A Shobhana
Flat 2A No. 17 Russa Road East First Lane Kolkata - 700 033, West Bengal; Institute of Neurosciences Kolkata, 185/1 A.J.C. Bose Road, Kolkata - 700 017, West Bengal
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.344620

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 » Abstract 


Melioidosis is an emerging tropical disease. Central nervous system (CNS) melioidosis may present as a brain abscess or encephalomyelitis mimics tuberculosis. Early diagnosis and management decreases mortality as well as morbidity. This case of brain abscess and encephalomyelitis in a young man with no known comorbidities was a diagnostic challenge. The surgery helped in debulking as well as isolating the causative organism. Appropriate antibiotic therapy for melioidosis was lifesaving and prevented further complications.


Keywords: Brain abscess, CNS infections, diagnosis, management, melioidosis
Key Message: Diagnosis of CNS infections is challenging. Melioidosis is one such infection that may have varied manifestations. A high index of suspicion and isolation of organisms is the cornerstone for appropriate management.


How to cite this article:
Shobhana A, Datta A, Trivedi S. CNS Melioidosis: A Diagnostic Challenge. Neurol India 2022;70:778-80

How to cite this URL:
Shobhana A, Datta A, Trivedi S. CNS Melioidosis: A Diagnostic Challenge. Neurol India [serial online] 2022 [cited 2022 May 22];70:778-80. Available from: https://www.neurologyindia.com/text.asp?2022/70/2/778/344620




Melioidosis, caused by Burkholderia pseudomallei, a motile, gram-negative bacillus is an emerging tropical disease in India.[1],[2] Central nervous system (CNS) melioidosis presenting as a primary cerebral abscess is rare[2] and reports are sparse due to difficulty in the diagnostic workup. Delayed diagnosis leads to high mortality. We report this rare case of CNS melioidosis presenting as both primary brain abscess and encephalomyelitis.


 » Case Report Top


A twenty-seven-year-old man was admitted in October 2019 with a low-grade fever of 3 weeks, headache and confusion of 5 days, and left-sided weakness of 3 days duration. He was admitted to a local hospital with one episode of generalized tonic-clonic convulsion. A contrast Computerized Tomography (CT) scan of the head showed a ring-enhancing lesion in the right temporoparietal lobe with significant perilesional edema. He was discharged on antitubercular drugs (ATT). He lived in a rural area, was unemployed, and apart from occasional alcohol intake, did not have any comorbidity.

On arrival at our hospital, he was very drowsy, with dense left hemiplegia. He had extensive Tinea corporis and cruris which raised suspicion of immunosuppression. Routine blood investigations were normal except for a white blood counts (WBCs) of 16,300/mL with neutrophil predominance and a serum Alanine aminotransferase (ALT) of 110 IU/L.

Magnetic Resonance Imaging (MRI) brain with contrast [Figure 1] revealed multiple well-defined ring-enhancing lesions of varying sizes in bilateral (right > left) cerebral hemispheres with mass effect. Spectroscopy favored infective etiology. Injection Ceftriaxone was started. The next day, his drowsiness increased necessitating intubation and ventilation. A guarded lumbar puncture was done. Cerebrospinal Fluid (CSF) revealed 35 cells (N40%, L60%), sugar 44 mg/dL, protein 115 mg/dL. Craniotomy and excision of the right temporal abscess were done. Sensorium remained poor for the next few days. His extraocular movements were restricted along with left Lower motor neuron (LMN) facial palsy. There were gram-negative bacilli on the Gram stain of pus and tissue. Zeihl Neelsen (ZN) stain, Periodic Acid Schiff (PAS) stains were negative. CSF and tissue  Mycobacterium tuberculosis Scientific Name Search TB) gene Xpert were negative. Culture grew Burkholderia pseudomallei. Histopathology of the right temporal lesion showed an organized pyogenic abscess [Figure 2]. ATT was stopped. Ceftriaxone was escalated to meropenem and IV Cotrimoxazole. He steadily improved. Parenteral antibiotics were continued for 6 weeks after which he was discharged on oral cotrimoxazole. Later, the family provided an additional history of his abuse of dexamethasone tablets for muscle building.
Figure 1: Contrast-enhanced MRI of the brain showing multiple well-defined ring-enhancing lesions of varying sizes are noted in bilateral (right > left) cerebral hemispheres, right ganglio-capsulo-thalamic region, superior cerebellar vermis, and upper cervical cord. Perilesional edema is noted adjacent to the right fronto-temporoparietal and ganglio-capsular lesions extending to the pons and the cervical cord (arrow)

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Figure 2: Photomicrograph of the right temporal lobe abscess. Gram stain of biopsy showing gram-negative bacilli with bipolar staining safety pin appearance (arrow) (X100 original magnification)

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 » Discussion Top


Melioidosis is underdiagnosed and underreported in India.[1],[2],[3] Agricultural, construction workers as well as immunocompromised individuals are at risk.[3] Based on the route of entry, melioidosis has various clinical manifestations. CNS involvement has been reported to be anywhere between 1.5 and 10%[2],[3],[4],[5],[6],[7] with a high mortality rate of up to 60%.[3],[4],[7] It is easily treatable but relapses and recurrences are common.[1],[6] A mimicker, Burkholderia can involve any organ of the body.[7] Radiologically and histopathologically, it resembles tuberculosis, hence, most often erroneously diagnosed and treated as tuberculosis.[6] Other differentials are  Brucellosis More Details, mycobacteria other than tuberculosis, fungi, and neoplastic lesions.[6] Brain stem encephalitis due to Listeria, Herpes, or Mycobacteria may have similar findings. Spread to the skin, bones, epidural and subdural space may be similar to tuberculosis, fungal infection, or malignancy. Hence, the diagnostic dilemma. A high index of suspicion is required for an accurate diagnosis. The presentation may be acute (symptoms lasting less than 2 months) or chronic.[1] While encephalomyelitis (37%) and brain abscess (35%) are common, scalp abscess and skull osteomyelitis may be seen in trauma cases. Brain abscess presents as fever and unilateral weakness. Fever and cranial nerve involvement, namely the seventh nerve along with the sixth, ninth, and tenth nerves are prominent in encephalomyelitis,[3] a characteristic of melioidosis. Paraplegia and quadriplegias have also been reported. Brain imaging and CSF are vital apart from blood cultures. In melioidosis, the CSF profile mostly showed mononuclear pleocytosis, high protein, and normal glucose.[3] The ring-enhancing pattern (78%) is the most frequent neuroimaging finding in encephalomyelitis and brain abscess. This pattern is seen in other infectious and inflammatory granulomatous lesions, demyelination, and neoplasms including metastases.[8] The brainstem (34%), frontal lobe (34%), and parietal lobe (33%) are commonly affected.[3] Diagnosis is clinched by isolating the organism mostly from brain tissue, pus, CSF, or blood (almost 50% of the cases). Serology (indirect hemagglutination assay) may help at times. With modern imaging and stereotactic techniques, even small abscesses of 1 cm diameter are amenable to aspiration.[9] Biopsy shows chronic abscess with focal granulomatous reaction mimicking tuberculosis, but non-AFB, non-spore bearing, bipolar staining—safety pin appearance helps to differentiate it from tuberculosis.[2] It is important to distinguish it from other gram-negative bacteria like Pseudomonas. Automated microbial identification systems generally do not identify this organism to the species level. All cases of melioidosis should be treated with initial intensive parenteral therapy of 2–8 weeks[1],[2],[3] with ceftazidime, meropenem, or imipenem. Meropenem is preferred due to fewer neurological side effects. For non-pulmonary sites of infection, the addition of trimethoprim-sulfamethoxazole (TMP-SMX) to ceftazidime or meropenem during the initial intensive therapy is recommended followed by an eradication phase of 3–6 months with oral TMP-SMX or doxycycline.

Our patient hails from rural Bengal. Presenting features of fever, neurological deficits, cranial nerve palsies along with mononuclear CSF pleocytosis in a patient from an endemic area with some risk factor for melioidosis should raise suspicion of this differential. We could convince the family for craniotomy which helped in diagnosis and source control. Antibiotics were appropriately escalated due to the critical condition of the patient. He is on maintenance oral TMP-SMZ now. Neurologically, his left hemiparesis is improving steadily with rehabilitation.


 » Conclusion Top


This case was a diagnostic challenge. Instead of assuming a common diagnosis of tuberculosis and waiting for an empirical ATT response, a high index of suspicion, timely surgery, isolation of the organism, and correct management of this tuberculosis-like presentation saved this young man from this life-threatening CNS infection.

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.
Karunarathna AKTM, Mendis SA, Perera WPDP, Patabendige G, Pallewatte AS, Kulatunga A, et al. A case report of melioidosis complicated by infective sacroiliitis in Sri Lanka. Trop Dis Travel Med Vaccines 2018;4:12.  Back to cited text no. 1
    
2.
Ganapathy S, Nair R, Kumar V. Rare presentations of CNS melioidosis - An institutional experience. J Neuroinfect Dis 2018;9:277.  Back to cited text no. 2
    
3.
Wongwandee M, Linasmita P. Central nervous system melioidosis: A systematic review of individual participant data of case reports and case series. PLoS Negl Trop Dis 2019;13:e0007320.  Back to cited text no. 3
    
4.
Pande A, Senthur Nambi P, Pandian S, Subramanian S, Ghosh S. Melioidosis mimicking tuberculous vertebral osteitis: Case report and review of literature. Neurol India 2018;66:1100-5.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Amarasena LP, Silva HDS, Tilakaratna PMYI, Jayamanne SF, Ranawaka UK. Melioidosis with a subdural collection – A case report. BMC Infect Dis 2019;19:143.  Back to cited text no. 5
    
6.
Madi D, Rai SP, Vidyalakshmi K, Chowta KN. Neurological melioidosis presenting as an intracranial abscess. Indian J Pathol Microbiol 2016;59:417-9.  Back to cited text no. 6
[PUBMED]  [Full text]  
7.
Chen GB, Tuan SH, Chen LH, Lin WS. Neurological melioidosis (Burkholderia pseudomallei) in a chronic psychotic patient treated with antipsychotics. A case report. Medicine 2018;97:e11110.  Back to cited text no. 7
    
8.
Haimes AB, Zimmerman RD, Morgello S, Weingarten K, Becker RD, Jennis R, et al. MR imaging of brain abscesses. AJR Am J Roentgenol 1989;152:1073-85.  Back to cited text no. 8
    
9.
Brouwer MC, Tunkel AR, McKhann GM, van de Beek D. Brain Abscess. N Engl J Med 2014;371:447-56.  Back to cited text no. 9
    


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