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We herein describe the case of an 18-year-old girl who presented with dizziness and headache in 2012. In 2013, brain magnetic resonance imaging revealed multiple intracerebral small lesions and intracerebral hydrocephalus. She was diagnosed with neurosarcoidosis following a brain biopsy. Although prednisolone, methotrexate, and azathioprine were administered, her hydrocephalus worsened and her granulomatous lesions were observed to increase in number on MRI. The patient's hydrocephalus showed no improvement despite her undergoing one ventriculoperitoneal shunt procedure, one septum pellucidum fenestration, and three ventriculoatrial shunt procedures. Infliximab therapy was then initiated, which resulted in a reduction in the size of the granulomatous lesions and the improvement of the patient's clinical symptoms. Infliximab may be a viable therapeutic option for treating intractable neurosarcoidosis.

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