Introduction: Neuroschistosomiasis is an uncommon and under diagnosed disease in our country because of the no clinical suspicion. The most common neurological manifestations are epileptic seizures as central nervous system involvement or different types of myelopathies: transverse myelitis, myeloradiculopathy, cauda equina syndrome or Brown-Sequard syndrome.

Case Report: A 27 years-old male from an endemic area, with atypical neurological affectation as he presented myelopathy and multifocal neuritis. Diagnosis was based on the epidemiological exposure datums, the myelopathy, the positive serological studies for Schistosoma haematobium, no detection of other parasitic infections and the clinical and radiological improvement after treatment. Cervical and thoracic magnetic resonance showed areas of hyper signal in T2 as it was described in other cases. It was detected S. haematobium in the bladder, the rest of serological and microbiological studies were negative. Besides, eosinophils on the biopsy of sural nerve orientative to parasitic etiology.

Conclusion: In patient with myelopathy or another unexplained neurological manifestation we have to suspect neuroschistosomiasis. In a world where migrations and travels are so frequent we have to think in this type of diseases.

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