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Neurology India, Vol. 58, No. 5, September-October, 2010, pp. 801-802 Letter to Editor Cerebral abscess and calvarial osteomyelitis due to Burkholderia pseudomallei Kalyan Bommakanti1, Praveen Ankathi1, P Uma2, Subbalaxmi Malladi3, V Laxmi2 1 Department of Neurosurgery, Nizam's Institute of Medical Sciences, Punjagutta, Hyderabad - 500 082, India Correspondence Address: Date of Acceptance: 19-Jul-2010 Code Number: ni10226 PMID: 21045524 DOI: 10.4103/0028-3886.72188 Sir, A 52-year-old paddy farmer presented with fever and abdominal pain. He had positive Widal titers and was started on parenteral ceftriaxone. He did not respond and developed headache, became drowsy and had two episodes of seizures. A computerized tomograph (CT) scan of the head [Figure - 1] showed ring-enhancing lesions with extensive perilesional edema in bilateral posterior parietal regions. Magnetic resonance imaging (MRI) of the brain [Figure - 2] showed ill-defined areas of altered signal intensity in bilateral posterior parietal regions, which were hypointense on T1-weighted (T1W) images, heterogeneously hyperintense on T2-weighted (T2W) images and with gyral enhancement. Extra-axial collections with peripheral rim enhancement were seen superficial to the sagittal sinus and in both parietal convexities. The overlying calvarium had altered signal intensity. The abscess was surgically evacuated. Intraoperatively, pus was seen under the scalp and osteomyelitis of the skull bones. Pus collections were also seen over the dura in bilateral parietal regions and also superficial to the superior sagittal sinus. Dura was breached and pus was seen tracking into brain parenchyma, forming cerebral abscesses. Pus under the scalp was drained. Osteomyelitic bone was removed. As the abscesses were on the sensori-motor area, total excision was not attempted. Pus was drained and capsule was partially removed. Burkholderia pseudomallei was isolated from both the blood and the pus cultures. The isolate was sensitive to cotrimoxazole, ceftazidime, imipenem and meropenem. Based on the microbiology report, the patient was put on parenteral ceftazidime (40 mg/kg) for two weeks followed by oral cotrimoxazole (8/40 mg/kg) for six months. The patient showed a rapid clinical improvement postoperatively and was asymptomatic at the third month of follow-up. B. pseudomallei is a saprophyte found in the soil and surface waters of rice fields. It is endemic in Northern Australia and South-East Asia. Although the Indian subcontinent is not an endemic region for Burkholderia, several cases of meliodosis have been reported. However, cases of central nervous system (CNS) melioidosis from India are sparse. [1] It spreads by inhalation and percutaneously through skin abrasions. Minor wounds and abrasions are common in farmers during the planting season and inoculation through these wounds might be the most common mode of spread. The incidence of CNS involvement in melioidosis ranges from 3% to 10%. [2],[3] It can manifest as focal encephalitis, encephalomyelitis, abscess, primary meningitis or osteomyelitis. [1] Skull osteomyelitis is either due to hematogeneous or contiguous spread and, secondarily, it leads to intracranial and extracranial abscess formation. Cerebrospinal fluid (CSF) analysis shows mononuclear pleocytosis, similar to tuberculosis. Encephalomyelitis can mimic Guillain-Barre syndrome. [4] Because the infection is associated with a high mortality, there is a need for an early and definitive diagnosis. MRI is far more sensitive than CT scan in the early stages, and a definitive diagnosis can be established by culture. Melioidosis can cause a false-positive Widal test. [5] It also shows partial response to ceftriaxone, but soon relapses. On histopathological examination, melioidosis shows chronic abscess with focal granulomatous reaction mimicking tuberculosis. The treatment protocol consists of an intensive phase with parenteral antibiotics for two weeks followed by a maintenance phase with oral antibiotics for 3-6 months. Ceftazidime crosses the blood brain barrier and is the drug of choice for CNS melioidosis. For strains resistant to ceftazidime, carbapenems are shown to have good activity. Ceftazidime (40 mg/kg) or imipenem (20 mg/kg) every 8-hourly is advised for a minimum duration of 10 days. [6] For the eradication phase, Currie et al.[2] recommend trimethoprim/sulfamethoxazole (TMP-SMX) at 8/40 mg/kg (up to 320/1,600 mg) every 12 h for 3-6 months. In India, melioidosis can mimic endemic infections like typhoid and tuberculosis clinically, radiologically and pathologically. There is a need for a high degree of clinical suspicion in patients with history of contact with surface water (paddy fields), recent travel to endemic regions and diabetes, who did not respond to routine antibiotics or anti-Kochs regimen. To prevent a fatal outcome, management should be aggressive with relevant surgical procedures and an intensive antibiotic regimen. Acknowledgment The authors would like to acknowledge Dr. Sreedhar Narayan, Dr. Harshavardhan and Dr. Srikanth for their help in preparing the manuscript and collecting the data. References
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