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PURPOSE To investigate the corneal endothelium damage in Graves ophthalmopathy (GO) and its role as a promising quantitative index to evaluate GO activity. DESIGN Cross-sectional study. METHODS This study included 128 eyes of 64 patients with GO. All subjects underwent ophthalmologic examinations, including proptosis, tear break-up time (BUT), corneal fluorescein staining, Schirmer test. Corneal endothelium was measured by noncontact specular microscope and ocular biometric parameters were measured by IOL Master 700. Each eye was assigned with a specific clinical activity score (CAS), then grouped as Active (CAS ≥ 3 points) or Inactive (CAS<3 points). Ocular parameters between the two groups were compared using generalized estimating equations accounting for intereye correlation, and receiver operating characteristic (ROC) curves were also obtained. MAIN OUTCOME MEASURES Parameters of corneal endothelium. RESULTS Among the included eyes, 81 eyes had inactive GO and 47 eyes had active GO. Corneal endothelial cell morphology was altered in active GO compared with inactive GO. The coefficient variation of cell area (CV) was significantly higher in active GO compared with inactive GO (37.0(34.4-41.2)% vs. 33.9(30.9-36.8)%, P=0.001), and positively correlated with CAS (r=0.322, P<0.001). Moreover, CV showed a diagnostic capacity to differentiate the active eyes from inactive eyes. The area under the ROC curve (AUC) was 0.705. CONCLUSIONS Active GO had morphological changes in corneal endothelium compared with inactive GO. CV is a sensitive indicator reflected corneal endothelial function, and has the potential to be adapted as a non-invasive, objective, and quantitative index for evaluating the activity status of GO patients.