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LETTERS TO EDITOR
Year : 2019  |  Volume : 67  |  Issue : 4  |  Page : 1149-1152

Isolated Cerebellar Abscess by Burkholderia pseudomallei in an Immunocompromised Host: A Rare Case


1 Department of Microbiology, AIIMS, Bhubaneswar, Odisha, India
2 Department of Neurosurgery, AIIMS, Bhubaneswar, Odisha, India
3 Department of Pathology and Lab Medicine, AIIMS, Bhubaneswar, Odisha, India

Date of Web Publication10-Sep-2019

Correspondence Address:
Dr. Ashoka Mahapatra
Department of Microbiology, AIIMS, Bhubaneswar, Odisha
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.266238

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How to cite this article:
Mahapatra A, Dhal S, Bansal S, Turuk J, Choudhury S, Mishra P. Isolated Cerebellar Abscess by Burkholderia pseudomallei in an Immunocompromised Host: A Rare Case. Neurol India 2019;67:1149-52

How to cite this URL:
Mahapatra A, Dhal S, Bansal S, Turuk J, Choudhury S, Mishra P. Isolated Cerebellar Abscess by Burkholderia pseudomallei in an Immunocompromised Host: A Rare Case. Neurol India [serial online] 2019 [cited 2019 Sep 23];67:1149-52. Available from: http://www.neurologyindia.com/text.asp?2019/67/4/1149/266238




Sir,

Burkholderia pseudomallei is a Gram-negative bacilli found in soil and surface water. This organism is endemic to the tropics, particularly north eastern Thailand and northern Australia, where the mortality rates approach 50% and 14%, respectively.[1] Virtually any organ of the body may be infected but most often involved are skin, soft tissues, lungs, liver, and rarely can infect the parotid gland, brain, and bone.

A 68-year-old female was brought to our hospital with the complaints of fever for 20 days and altered sensorium of 1 day. On examination, she was conscious but disoriented with a GCS score (Glasgow Coma Scale) of E4V4M6 with no motor weakness, febrile (39°C), pulse – 80 beats/min, and BP – 114/70 mm Hg. She was a known case of type 2 diabetes mellitus. Other systemic examination revealed no abnormality.

Initial brain computed tomography (CT) was performed and showed left cerebellar hypodensity [Figure 1]a. On subsequent magnetic resonance imaging (MRI) brain with contrast, there was a ring enhancing left cerebellar lesion with surrounding edema with no hydrocephalus [Figure 1]b. With these findings, the patient was diagnosed to be a case of left cerebellar abscess and after written and informed consent taken for surgery. Midline suboccipital craniotomy and gross total excision (GTE) of left cerebellar abscess under general anesthesia (GA) was done. Postoperative CT showed complete excision of left cerebellar lesion [Figure 1]c. Excised abscess material was sent for biopsy and aerobic and anaerobic culture. Biopsy report revealed abscess with secondary changes [Figure 2]. Her investigation reports were – random plasma glucose – 237 mg/dl, HbA1C – 7.3%, TLC – 8.4 × 103/cmm, Hb – 9.8 g/dl, urea – 25 mg/dl, creatinine – 1.03 mg/dl, sodium – 142 μg/L, potassium – 2 μg/L, calcium 8 mg/dl, and urine for acetone was negative. Empirical intravenous antibiotics (ceftriaxone, amikacin, and metronidazole) as per protocol were given. The pus culture report was received after 72 h, which showed no growth on anaerobic culture. But aerobic culture showed growth on blood agar and non-lactose fermenting colonies on MacConkey agar. They were Gram-negative bipolar staining bacilli, motile, catalase, oxidase positive, produced an alkaline slant and butt without hydrogen sulfide on triple sugar iron agar, negative results for indole, methyl red, and Voges–Proskauer test, and positive for nitrate reduction and citrate utilization. It also oxidized arabinose with other routine sugars. The organism was identified as B. pseudomallei and complete result was reported on the 4th day after the operation. The organism was sensitive to carbapenems, ceftazidime, ciprofloxacin, cotrimoxazole, and tigecycline and resistant to aminoglycosides, colistin, and polymixin B [Figure 3]. Two samples of blood were also received for culture, but reported sterile after 7 days. Serology for B. pseudomallei (agglutination assay) could not be performed. The medical treatment was switched to Inj. ceftazidime up to 2 weeks of operation. The patient's clinical condition improved with given treatment. The patient was discharged with advice of maintenance treatment with co-trimoxazole for 6 months with follow-up advice of monthly follow-up. The patient was found to be all right without any symptoms on follow-up visits.
Figure 1: Non-contrast computed tomography head showing left cerebellar hypodense mass (a), contrast magnetic resonance imaging brain showing left cerebellar ring enhancing lesion (b), postoperative computed tomography showing excision of left cerebellar lesion (c)

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Figure 2: Photomicrograph (H and E, 2×) showing fragments of cerebellar tissue with mixed inflammatory exudates

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Figure 3: Antibiogram of B. pseudomalleii

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B. pseudomallei, the causative agent of melioidosis is considered as one of the potential biological warfare agent. Melioidosis is considered to be underreported and an emerging disease worldwide. Thailand has reported the largest number of cases of melioidosis, estimated to be 2000–3000 each year. The first case of melioidosis from India was reported in a child from Dapolitaluka in Maharashtra in 1990. Subsequently several cases have been reported from Karnataka, Tamil Nadu, Kerala, and Maharashtra.[2] B. pseudomallei are easily isolated from irrigated rice paddies, clay soils, and standing water from endemic regions and human infection is closely associated with high rainfall. Percutaneous inoculation and inhalation are thought to be the two common routes of transmission and inhalation of contaminated water droplets or dust (soil) is probably the most important natural route of infection. The olfactory nerve fascicles underneath the epithelium coalesce to form larger bundles that project through the bony cribriform plate to enter the central nervous system. The nasal mucosa is also innervated by the ophthalmic branch of the trigeminal nerve, which could also provide a direct route to the brain, and is of concern to neurological melioidosis. This disorder could be seen at any age with a male preponderance. However, the present case is an elderly female with no such specific history. Diabetes mellitus, alcoholism, renal disease, immunosuppression, and thalassemia are the widely documented risk factors for melioidosis. Ours was a known case of type II diabetes mellitus. The clinical manifestations of melioidosis are ranging from chronic abscess formation to fulminant sepsis. This can be classified into four clinical groups; disseminated septicemic, non-disseminated septicemia, localized, and multifocal localized. Disseminated septicemic melioidosis is defined as positive blood culture and more than one organ involved; non-disseminated septicemic is defined as positive blood culture and only one foci or no apparent focus of infection; localized melioidosis is defined as only single focus of infection with negative blood culture; and multifocal localized is defined as multiple organs involvement but negative blood culture.[3] Melioidosis is accepted as a great mimicry pathogen as the clinical symptoms mimic those of many other diseases. The presenting symptoms may vary from fever, dry cough due to irritation of diaphragm by abscess, abdominal pain, localized swelling to septicemia, and shock. Lung is the most commonly involved organ in melioidosis. Abscess formation in multiple internal organs such as the liver and spleen is common in immunocompromised hosts. But our case presented with isolated cerebellar abscess without any other organ involvement even though she was having a risk factor of type II diabetes mellitus that is rarely been reported previously [4],[5],[6],[7],[8],[9],[10],[11],[12],[13] [Table 1]. It is well documented that the clinical and radiologic signs of neurologic melioidosis can mimic those of neurologic tuberculosis and viral encephalitis. The definitive diagnosis is always isolation of the organism from the specimen. The diagnosis in our case was confirmed by a positive culture of B. pseudomallei from the aspirated pus of cerebellar abscess. It is difficult to differentiate B. pseudomallei from other Gram-negative oxidase positive motile bacilli especially pseudomonas. Direct immunofluorescence microscopy is 98% specific and 70% sensitive compared to culture and an indirect hemagglutination assay (IHA) is only 56% sensitive for the detection of melioidosis.[14] B. pseudomallei is characteristically resistant to penicillin other than ureidopenicillins, ampicillin, first- and second-generation cephalosporins, aminoglycosides, polymixin B, colistin, and macrolides. It is only susceptible to chloramphenicol, tetracyclines, co-trimoxazole, ureidopenicillins, third-generation cephalosporins, carbapenems, and amoxicillin–clavulanate. The antibiotic of choice for melioidosis is ceftazidime, which is found to be more effective than other third generation cepaholsporins. Intravenous ceftazidime or imipenem is usually administered for the eradication phase followed by oral maintenance therapy with either co-trimoxazole or doxycycline for at least 6–8 months. Reduction in mortality rate has been reported by using a combination of ceftazidime and co-trimoxazole in a randomized trial.[15] Recently Kandasamy and Norton have reported ceftazidime or meropenem alone or with the addition of co-trimoxazole as the most active regimen.[16]
Table 1: Published Indian series on CNS melioidosis

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Infections due to B. pseudomallei is under diagnosed and underreported in India, may be due to a lesser frequency of suspicion among the clinicians as well as microbiologists. As the clinical features of neuromelioidosis are relatively non-specific, neuroimaging features should alert the clinician regarding its probability which can help for early microbiological sampling. Moreover the antimicrobial susceptibility pattern of this organism is different, so high degree of suspicion should be needed by the microbiologist for its identification, appropriate antibiogram, and duration of therapy.

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.
St John JA, Ekberg JA, Dando SJ, Meedeniya AC, Horton RE, Batzloff M, et al. Burkholderia pseudomallei penetrate the brain via destruction of the olfactory and trigeminal nerves: Implications for the pathogenesis of neurological melioidosis. MBio 2014;5:e00025.  Back to cited text no. 1
    
2.
Mukhopadhya A, Balaji V, Jesudason MV, Amte A, Jeyamani R, Kurian G. Isolated liver abscesses in melioidosis. Indian J Med Microbiol 2007;25:150-1.  Back to cited text no. 2
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3.
Dhiensiri T, Eua-Ananta Y. Visceral abscess in melioidosis. J Med Assoc Thai 1995;78:225-31.  Back to cited text no. 3
    
4.
Vidyalakshmi K, Shrikala B, Bharathi B, Suchitra U. Melioidosis: An under-diagnosed entity in western coastal India: A clinico-microbiological analysis. Indian J Med Microbiol 2007;25:245-8.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Mukhopadhyay C, Chawla K, Krishna S, Nagalakshmi N, Rao SP, Bairy I. Emergence of Burkholderia pseudomallei and pandrug-resistant non-fermenters from southern Karnataka, India. Trans R Soc Trop Med Hyg 2008;102:S12-7.  Back to cited text no. 5
    
6.
Kumar GS, Raj PM, Chacko G, Lalitha MK, Chako AG, Rajshekhar V. Cranial melioidosis presenting as a mass lesion or osteomyelitis. J Neurosurg 2008;108:243-7.  Back to cited text no. 6
    
7.
Antony B, Pinto H, Dias M, Shetty AP, Scaria B, Kurivilla T, et al. Spectrum of melioidosis in the suburbs of Mangalore, S West coast of India. Southeast Asian J Trop Med Public Health 2010;41:169-74.  Back to cited text no. 7
    
8.
Bommakanti K, Ankathi P, Uma P, Malladi S, Laxmi V. Cerebral abscess and calvarial osteomyelitis due to Burkholderia pseudomallei. Neurol India 2010;58:801-2.  Back to cited text no. 8
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9.
Naha K, Dasari S, Kusugodlu R, Prabhu M. Cranial melioidosis with extradural extension after a fall in the bathroom. Australas Med J 2012;5:455-8.  Back to cited text no. 9
    
10.
Garg G, Chawla N, Chawla K, Khosla P, Jain S. Atypical presentations of melioidosis in North India: Report of two cases. J Assoc Physicians India 2015;63:82-3.  Back to cited text no. 10
    
11.
Saravu K, Mukhopadhyay C, Vishwanath S, Valsalan R, Docherla M, Vandana KE, et al. Melioidosis in Southern India: Epidemiological and clinical profile. Southeast Asian J Trop Med Public Health 2010;41:401-9.  Back to cited text no. 11
    
12.
Shetty HS, Mallela AR, Shastry BA, Acharya V. Parietal bone osteomyelitis in melioidosis. BMJ Case Rep 2015;2015. doi: 10.1136/bcr-2014-208612.  Back to cited text no. 12
    
13.
Prasad GL, Nair RP, Menon GR. Intracranial melioidosis:First report in a human immunodeficiency virus positive individual manifesting as cranial osteomyelitis. Neurol India 2017;65:1423-6.  Back to cited text no. 13
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14.
Cheng AC, O'Brien M, Freeman K, Lum G, Currie BJ. Indirect hemagglutination assay in patients with melioidosis in northern Australia. Am J Trop Med Hyg 2006;74:330-4.  Back to cited text no. 14
    
15.
Thibault FM, Hernandez E, Vidal DR, Girardet M, Cavallo JD. Antibiotic susceptibility of 65 isolates of Burkholderia pseudomallei and Burkholderia mallei to 35 antimicrobial agents. J Antimicrob Chemother 2004;54:1134-8.  Back to cited text no. 15
    
16.
Kandasamy Y, Norton R. Paediatric melioidosis in North Queensland, Australia. J Paediatr Child Health 2008;44:706-8.  Back to cited text no. 16
    


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