Melioidosis of the nervous system: atypical presentation of a rare disease in a 48-year-old man

Abstract

A 48-year-old man who worked in mining in remote, northern Australia was transferred from a rural hospital 5 days after the onset of headaches, subjective fevers and flaccid paralysis of the left upper limb. Initial investigations demonstrated inflammatory cerebrospinal fluid (CSF) changes and a longitudinally extensive cervical cord lesion. Given two serial negative blood and CSF cultures, he was treated as inflammatory myelitis with intravenous methylprednisolone. Despite the initial improvement in pain and left arm power, the patient’s neurological deficit plateaued and then deteriorated with worsening neck pain, diaphragmatic dysfunction and dysphagia requiring intubation and respiratory support. A third CSF culture isolated Burkholderia pseudomallei confirming a diagnosis of neuro-melioidosis. Repeat imaging revealed the rostral extension of the original spinal cord lesion into the medulla and pons. Over the next 4 weeks, the patient’s neurological deficits slowly improved with continued intravenous antibiotic therapy with meropenem and oral trimethoprim/sulfamethoxazole.

Critical approach to atypical spectrum of melioidosis: a case-series based literature review

“…A study by Hamdoon et al outlined the neurological manifestations consistent with acute pyogenic meningitis such as fevers (76%), headaches (54%) and altered mental status (39%) as seen in our case above 20. Atypical displays of neuromelioidosis include cranial nerve palsies, focal neurological deficits, brainstem encephalitis and cerebellar involvement as per Padiglione et al ,20 21 and can also present as a stroke-like illness depending on foci of involvement 22. Furthermore, a case report by Arif et al demonstrates a man in his 30s with peripheral neuropathy secondary to melioidosis 23.…”

“…Neuromelioidosis constitutes only 3%–4% of total melioidosis cases with a significant debilitating morbidity and mortality rate reaching as high as 25%. A study by Hamdoon et al outlined the neurological manifestations consistent with acute pyogenic meningitis such as fevers (76%), headaches (54%) and altered mental status (39%) as seen in our case above 20. Atypical displays of neuromelioidosis include cranial nerve palsies, focal neurological deficits, brainstem encephalitis and cerebellar involvement as per Padiglione et al ,20 21 and can also present as a stroke-like illness depending on foci of involvement 22.…”

“…Furthermore, a case report by Arif et al demonstrates a man in his 30s with peripheral neuropathy secondary to melioidosis 23. Few other rare presentations of neuromelioidosis reported are cerebral/dural(sagittal) sinus thrombosis and mimicking acute transverse myelitis 20 24 25…”