During visceral leishmaniasis (VL), chronically infected dogs may present neurological disorders, nevertheless, the pathogenesis of the cerebral form was not still elucidated. Toll-like receptors (TLRs) recognize the lipophosphoglycan, present in the Leishmania parasite, and mediate the production of proinflammatory cytokines. The aim of this study was to evaluate the histopathological lesions and gene expression of TLRs and cytokines in the central nervous system from fifteen dogs with VL and four uninfected. Samples from brain (pool) and choroid plexi were collected in RNAlater® and others samples were subjected to routine histological procedures and staining with haematoxylin-eosin. RNA was extracted, reverse transcript to cDNA and submitted to RT-qPCR to quantify the gene expression of TLR-2, TLR-9, IL-1β and TNF-α, using specific primers and Taqman® probes. G3PDH was used as a reference gene. Meningitis and choroiditis were detected in the infected dogs. There was upregulation of TLR-2 gene expression in the choroid plexus (3.09-fold more; p = 0.047) and trend of upregulation in the brain from infected dogs (2.69-fold more; p = 0.0799), while TLR-9 presented no differences between groups in the two areas. Moreover, IL-1β was up-regulated (6.36-fold more; p < 0.001) along with TNF-α (4.31-fold more; p = 0.001) in the brain of infected dogs, while there was no differences in the choroid plexus between groups. This indicates that TLR-2, IL-1β and TNF-α may be produced in the brain, and TLR-2 in the choroid plexus, contributing to the development of the brain inflammatory process, contrasting to TLR-9, that seems not to be related to the brain inflammatory lesion.