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Background and Aim: Although endoscopic recognition of dysplasia in Barrett's esophagus is difficult, experience in recognition of early neoplastic lesions is supposed to increase the detection of early neoplastic lesions. The aim of this study was to assess the significance of dysplasia in random biopsies in Barrett's esophagus, in the absence of reported visible lesions as well as the difference in final outcome of pathology. Methods: We retrospectively identified all patients with Barrett's esophagus with suspicion of dysplasia or early adenocarcinoma who were referred to our center between February 2008 and April 2016. We analyzed all endoscopy reports, pathology reports, and referral letters from 19 different hospitals. Patients were divided into two groups, based on the presence or absence of visible lesions reported upon referral. Results: In total, 170 patients diagnosed with dysplasia or adenocarcinoma were referred to our tertiary center. Ninety-one of these referred patients were referred with dysplasia or adenocarcinoma in random biopsies, without a reported lesion during endoscopy in the referral center. During endoscopic work-up at our center, a visible lesion was detected in 44 of these 91 patients (48.4%). After endoscopic work-up and treatment, adenocarcinoma was found in an additional 21 patients. Two of these patients were initially referred with low-grade dysplasia, and 19 patients were initially referred with high-grade dysplasia. The final pathology was upstaged in 35.8% of the patients. Conclusions: The presence of any grade of dysplasia in random biopsies during surveillance in referral centers is a marker for more severe final pathology. Training in recognition of early neoplastic lesions in Barrett's esophagus imaging is recommended for endoscopists performing Barrett's surveillance. [ABSTRACT FROM AUTHOR]