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Background: Melioidosis caused by Burkholderia pseudomellei is an infection with protean clinical manifestations. Guillain-Barré syndrome [GBS] associated with melioidosis is very rare., Case Presentation: A 42-year-old woman with diabetes presented with abdominal pain, vomiting and intermittent fever for one month. Six months before presentation she had recurrent skin abscesses. Three months before presentation she had multiple liver abscesses which were aspirated in a local hospital. The aspirate grew "coliforms" resistant to gentamicin and sensitive to ceftazidime. On presentation she had high fever and tender hepatomegaly. Ultra Sound Scan of abdomen showed multiple liver and splenic abscesses. Based on the suggestive history and sensitivity pattern of the previous growth melioidosis was suspected and high dose meropenem was started. Antibodies to melioidin were raised at a titre of 1:10240. The growth from the aspirate of liver abscess was confirmed as Burkholderia pseudomellei by polymerase chain reaction [PCR]. After a week of treatment, patient developed bilateral lower limb weakness. Deep tendon reflexes were absent. There was no sensory loss or bladder/bowel involvement. Analysis of the cerebro-spinal fluid showed elevated proteins with no cells. There was severe peripheral neuropathy with axonal degeneration. A diagnosis of GBS was made and she was treated with plasmapharesis with marked improvement of neurological deficit. Continuation of intravenous antibiotics lead to further clinical improvement with normalization of inflammatory markers and resolution of liver and splenic abscess. Eradication therapy with oral co-trimoxazole and co-amoxyclav was started on the seventh week. Patient was discharged to outpatient clinic with a plan to continue combination of oral antibiotics for 12 weeks. At the end of 12 weeks she was well with complete neurological resolution and no evidence of a relapse., Conclusions: Guillaine Barre syndrome is a rare complication of melioidosis and should be suspected in a patient with melioidosis who develop lower limb weakness. Plasmapharesis can be successfully used to treat GBS associated with active melioidosis.