Human alveolar echinococcosis (AE) is a severe hepatic disease caused by the dog/fox tapeworm Echinococcus multilocularis. The parasite is cycling between rodents and foxes/dogs. AE has been considered as one of major health problems in Yili Valley, Xinjiang, China. A total 268 AE cases were found from 1989 to 2015 in the Valley with 64.3% of the patients diagnosed in the last 10 years. The most patients (76%) were aged from 21 to 55. Among the patients, 62% were nomad farmers, indicating that sheep-farming is a high risk position in the valley. The life-cycle of E. multilocularis may involve Kazakhstan voles (Microtus ilaeus), one of major rodents in Yili Valley. The prevalence of AE in this species ranged from 0.76% to 10.4%. The rodent had 0.78 burrow per m2 in the area close to stream or brook, whereas, 0.21 burrow per m2 in the area 200 meters away from the stream or brook. In average, one individual M. ilaeus had 16.2 burrow entrances linked with runways in the pasture area. The small mammals like to inhabit in the pasture land close to streams, where is the location for Kazak and Mongolian herdsmen setting their yurts during the summer season. Those herdsmen may have high risk for infection with AE. Dogs are important definitive host and play a key role in transmission of AE to humans in the valley. Control of alveolar echinococcosis should focus on controlling the population of Microtus voles, deworming dogs and educating villagers to increase the knowledge of prevention and control of echinococcosis.