Background: Neurological sequelae are a rare but recognised complication of Chikungunya infection. We report a case of acute haemorrhagic leukoencephalitis secondary to Chikungunya infection. Case description: A 59 year old male presented to the acute medical unit 5 days after returning from a 4 week trip to Kinghasa in the Democratic Republic of Congo complaining of a sudden onset headache with fluctuating confusion and left sided weakness. 1 week previously he had been complaining of fever and fatigue with arthralgia. Whilst in DRC he visited family with no travel outside Kinghasa and no infectious contacts or high risk activities. A malaria film was negative and a lumber puncture on admission demonstrated 296/mm3 white cells with 75% polymorphs and 25% lymphocytes, protein of 1.5 g/L and glucose of 4.2 mmol/L ( serum 7.4 mmol/L). Over the next 24 hours his GCS dropped and he needed intubation. The appearance of the MRI head was highly unusual and the neuroradiologist felt this was most likely an acute haemorrhagic leukoencephalitis (AHLE) confirmed later on brain biopsy. All subsequent investigations were negative except viral serology demonstrating a positive chikungunya IgM with later IgG seroconversion. The patient received steroids, anti tuberculous treatment, antibiotics and plasma exchange with little neurological improvement. Conclusion With the increasing incidence of chikungunya, more neurological complications are being recognised. AHLE is a very rare form of acute disseminated encephalomyelitis usually triggered by an infection; however there have been no case reports of chikungunya causing AHLE leading us to believe this may be the first case.