A comprehensive study of human echinococcosis (caused by Echinococcus granulosus or E. multilocularis), including assessment of hospital records, community surveys and patient follow-up, was conducted in Ningxia Hui Autonomous Region (NHAR), China. In contrast to hospital records that showed 96% of echinococcosis cases were caused by cystic echinococcosis (CE), 56% of cases detected in active community surveys were caused by alveolar echinococcosis (AE). The AE and CE cases co-existed frequently in the same village, even occurring in the same patient. A serious public health problem caused by echinococcosis was evident in southern NHAR, typified by: a long diagnostic history for both AE and CE (7.5 years) compared with a shorter treatment history (4.7 years); a significant mortality rate (39%) caused by AE in one surveyed village, where patients had no previous access to treatment; family aggregation of CE and AE cases; a high proportion of both AE (62.5%) and CE (58%) in females; a high rate of recurrent surgery (30%) for CE demonstrated by surgical records; and frequent symptomatic recurrences (51%) because of discontinuous or sporadic access to chemotherapy for AE. The disease burden for both human AE and CE is thus very severe among these rural communities in NHAR, and this study provides the first attempt to determine the costs of morbidity and surgical intervention of human CE and AE cases both at the hospital and community level in this setting. This information may be useful for assessing the cost effectiveness of designing effective public health programs to control echinococcosis in this and other endemic areas in China and elsewhere.