Hydatidosis is a zoonotic disease of worldwide distribution caused by Echinococcus granulosus. Our study aimed to determine the prevalence of human and canine echinococcosis as well as the associated risk factors in a rural area of the Limarí province in northern Chile. A cross-sectional study was conducted between August and November 2009 using a stratified sampling design in each of the five districts of the province. In the selected villages, up to 10 households were sampled. Serum and fecal samples from an adult family member and a dog were collected from each participating household. Risk factors were assessed by standardized questionnaires. Seroprevalence was assessed using a multi-step approach: an ELISA for screening, IFA, IHA and western blot for confirmation of results, respectively. The prevalence of echinococcal infection in dogs was determined by coproantigen genus specific ELISA. Chi-square, Fisher tests and logistic regressions were used to assess risk factors for human seropositivity and dog copropositivity. A seroprevalence of 2.6% (10/403) and coproprevalence of 28% (26/93) was recorded for humans and dogs respectively. Contact with dogs and dog feces were risk factors for human seropositivity while dog copropositivity was associated with home slaughter of livestock (OR = 3.35; CI 90%: 1.16–6.85) and households de-worming dogs (OR = 2.82; CI 90%: 1.33–8.43). Echinococcal infection of humans and their dogs is common in Limarí province. Risk factors for human seropositivity were related to contact with domestic dogs and their feces, whereas those for dogs were home slaughter of livestock and the practice of de-worming dogs. Hydatidosis is a hyperendemic zoonotic disease in Chile caused by the dog tapeworm, Echinococcus granulosus. In Chile as in many other countries in South America, this disease has been largely neglected with few exceptions. Chile's growing economy and the interest of health authorities has lead to an increase in the number of studies investigating the epidemiology of echinococcosis and the factors related to infections of the main definitive host, the domestic dog and humans. In this study, we determined the prevalence of human and canine echinococcosis as well as the associated risk factors in a rural area of the Limarí province in northern Chile. We undertook a household questionnaire survey in rural areas of the five municipalities of the Limarí province in Coquimbo region. For each household serum of an adult family member and fecal samples from a dog were taken. Results of our study indicate that infection occurs in 2.6% of humans and 28% of dogs and is primarily due to feeding of dogs with contaminated offal and high dog-human contact. As a result of this study, the Chilean Ministry of Health instigated a control program aimed to control the infection in dogs and avoid new infections to humans.