Neospora caninum is a cyst-forming apicomplexan protozoan parasite that infects canids as definitive and a wide range of mammals as intermediate hosts. The parasite is mostly known by its role in the abortion in cattle with a significant economic loss however, serological evidences suggest the infection of the one-humped camel (Camelus dromedarius). The aim of this study was to employ molecular and histopathological methods to examine N. caninum infection in blood and tissues from 100 camels in Iran. Genomic DNA was extracted from blood, brain, liver and portal lymph node specimens of the camels, and nested-PCRs targeting ITS-1 region of N. caninum (NN1, NN2, NP1, NP2 primers) were performed. In addition, brain tissue samples were fixed in 10% neutral buffered formalin and embedded in paraffin. Sections (5 μm) were cut, stained with haematoxyline and eosin (H&E), and examined microscopically. Results revealed the DNA of N. caninum in the blood, brain and portal lymph nodes of two (2%) camels. Histopathology also revealed cysts resembling N. caninum in brain samples of PCR- positive camels. This study provides the first insight into detection of N. caninum in Camelus dromedarius. Further molecular studies specifically on the fetal tissues are required for better understanding of the epidemiology and transmission dynamics of N. caninum in camels.