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While small solitary oesophageal papillomas are uncommon findings that are occasionally identified on routine upper endoscopies, extensive oesophageal papillomatosis is an extremely rare condition with only 9 cases reported in the English literature. We report the case of a 72-year-old woman who was referred for progressive dysphagia for solid food and clinical signs for a reflux disease. Upper endoscopy demonstrated bizarre villous alterations of the mucosa covering the oesophagus subtotally and a suspicious area within these alterations. Histological work-up of the biopsy samples revealed marked papillary hypertrophy and a squamous epithelial carcinoma in situ corresponding to the suspicious lesion. The patient underwent oesophagectomy with cervical gastroesophageal anastomosis and proximal remnants of papillomatous mucosa above the anastomosis were destroyed with endoscopic argon plasma coagulation. In the 2-year follow-up the patient showed limited recurrence of the papillomatosis in the remaining proximal oesophagus containing a circumscript carcinoma that was successfully treated by local endoscopic mucosectomy. Our case strongly underscores the risk of malignant transformation in large areas of papillomatous mucosa and shows that systematic surveillance is essential.