Objective To understand the status of infection and knowledge of human echinococcosis prevention in Maqin County of Qinghai Province. Methods A cluster random sampling method was used to select permanent residents at age over 2 years from 6 townships and 2 towns in Maqin County for B-ultrasound examination during 2017-2018 to calculate the prevalence rate of echinococcosis in the population. A questionnaire survey was performed randomly among local residents and schoolchildren of fourth grade and higher to estimate the awareness rate of knowledge on echinococcosis control. The estimated rates were compared using Chi-square test. Results A total of 36 705 residents were examined by B ultrasound scanning, and 433 were found of echinococcosis, with a prevalence rate of 1.18% (433/36 705). In 2017 and 2018, 485 and 436 questionnaires were distributed, respectively, which revealed awareness rate of echinococcosis knowledge of 89.69% and 91.74%, respectively. The prevalence of echinococcosis in females (1.58%, 280/17 746) was higher than that in males (0.81%, 153/18 959) (P < 0.01). The prevalence showed a trend of increase with age, with the age group of ≥70 years being the highest (2.63%, 38/1 446). Among all occupation groups, the prevalence rate was highest (2.56%) in herdsmen, and lowest (0.06%) in officials. Among educational levels, the echinococcosis prevalence was highest (3.15%, 235/7 466) in the illiterates, followed by the primary school level (1.30%, 183/14 033), and the lowest in group with the university education (0.03%, 2/5 995). As to the residing patterns, the nomads had the highest prevalence of echinococcosis (4.67%, 107/2 290). The prevalence was found highest in Youyun Township (7.67%, 95/1 238). It was noted that the prevalence difference between males and females, age groups, occupations, educational levels, residing patterns and geographical locations was statistically significant (P < 0.05). In 2017 and 2018, the knowledge awareness rate of echinococcosis prevention and control was lowest in herdsmen (66.33%, 65/98; 79.34%, 96/121), and highest in officials (98.89%, 89/90; 98.77%, 80/81) among the occupations; and lowest in the education group of primary school (78.77%, 141/179; 86.08%, 167/194), and highest in the undergraduates (98.80%, 90/91; 98.77%, 80/81). There were significant differences in the awareness rate of knowledge of echinococcosis prevention and control among different occupations and education levels (P < 0.05). Conclusion The situation of echinococcosis control in Maqin County is still challenging, showing a prevalence rate of 1.18%. Although the knowledge awareness rate on echinococcosis has been improved, it is essential to strengthen health education, particularly for the key target population of herdsmen.