Human brucellosis represents a serious public health concern in Hulun Buir and requires a comprehensive epidemiologic analysis to define adapted control measures. The present study describes the case numbers, constituent ratios and incidence rate of human brucellosis. Conventional biotyping, that is abortus, melitensis, ovis and suis (AMOS)‐PCR and multi‐locus variable‐number tandem repeat analysis (MLVA) were used to characterize the Brucella strains. Between 2003 and 2018, a total of 23,897 human brucellosis cases were reported, with an incidence rate of 56.03/100,000, which is 20 times higher than the country's average incidence. This incidence rate increased year after year, culminating in 2005 and decreased between 2011 and 2018. Because Hulun Buir relies on a nomadic livestock rearing system, brucellosis spreads easily among different animal species and humans. In Xin Barag Left Banner and Xin Barag Right Banner, the incidence rates were, respectively, 226.54/100,000 and 199.10/100,000, exceeding those observed in other areas. Most of the cases occurred in the 25‐ to 45‐year‐old group, accounting for 65.74% of the cases (15,709/23,897), and among farmers, accounting for 66.71% (15,942/23,897). The male to female incidence ratio was 2.67:1. The higher incidence in younger people and the large gender ratio reflected the unique traditional production and lifestyle of nomads. Most reported cases were observed from April to June, indicating that more than 40% of the cases were related to the delivery of domestic livestock. The biotyping showed that the 44 isolated strains were all B. melitensis, including 12 Brucella melitensis biovar (bv) 1 and 32 B. melitensis bv. 3. The strains displayed a genetic similarity of 80%–100%. Our hypothesis is that human brucellosis outbreak in this region may be originating from a limited source of infection, so further investigation is necessary. The epidemic situation of human brucellosis in Hulun Buir is extremely serious, strengthened surveillance and control in animals’ brucellosis should be priority.