Neurobrucellosis is a frequent infection in the Mediterranean countries but it is under-diagnosed because of its clinical polymorphism. Clinical manifestations are very different in relation to meningeal, parenchymal or vascular involvement. Isolated intracranial hypertension is a rare symptom.

We report the case of a young man with neurobrucellosis who presented with isolated intracranial hypertension without other systemic symptoms of brucellosis.

A 32-year-old man with no medical history was referred to our department for headache and binocular diplopia evolving for 1 month. There was no history of drug use, consumption of raw milk, or weight gain or loss. Clinical examination showed bilateral stage II papilledema with a left VI nerve palsy. The patient had no meningeal syndrome or organomegaly. There was no fever. An intracranial hypertension was suspected and the patient was hospitalized. Cerebral magnetic resonance imaging was normal. The opening cerebrospinal fluid (CSF) pressure was 45 cmH₂O. Microscopic examination of the CSF revealed 113 cells/mL with lymphocytic predominance and negative culture. Protein level was 1.19 g/l, glucose level was 0.54 mmol/l (ratio at 0.09). Wright test was positive at 1/5120 titer in serum. The patient was diagnosed with neurobrucellosis and was treated with Doxycycline and Rifampicin with good clinical evolution.

The incidence of neurobrucellosis ranges between 1.7-10%. Chronic meningitis with intracranial hypertension has been rarely described as the initial manifestation of neurobrucellosis. The evolution is favorable in more than 50% of cases after appropriate antibiotic therapy. Early diagnosis and appropriate management are important.