Leptospirosis is a highly prevalent, globally distributed zoonosis with an incidence 10 times higher in tropical regions, where the proportion of new cases correlates with rainy seasons. Secondary effects to the central nervous system in the immune phase of the illness typically manifest as aseptic meningitis. However, neurologic sequelae can be diverse and, although rare, other central and peripheral neurologic presentations have been described including the Guillain-Barré syndrome. Neuroleptospirosis as a primary disease manifestation is even more unusual. Below, we present the case of a 27-year-old woman with a Guillain-Barré–like syndrome as a primary manifestation of neuroleptospirosis as confirmed by positive IgM serology test result in paired samples and microscopic agglutination test reactive for Leptospira icterohaemorrhagiae in the convalescent phase. The patient had a favorable clinical outcome after treatment with antibiotics, corticosteroids, ventilator support, and physical therapy.