Diffuse xanthoma in early esophageal cancer: A case report

BACKGROUND: Gastrointestinal xanthomas are asymptomatic and infrequent non-neoplastic lesions that commonly occur in the stomach with Helicobacter pylori-associated gastritis and rarely in the esophagus. To date, there have been no reports of esophageal xanthoma combined with esophageal cancer. Herein, we present the first case in the literature of a diffuse xanthoma complicated with early esophageal cancer. Moreover, this combination makes the endoscopic diagnosis difficult if it is not in mind. CASE SUMMARY: A 68-year-old man visited our department with a 2-mo history of epigastric discomfort. He underwent surgery for gastric cancer 6 years ago. Esophagogastroduodenoscopy showed a semi-circumferential irregular yellowish-colored and granular lesion in the esophagus (30-35 cm from the incisors). Using magnifying endoscopy with narrow band imaging, aggregated minute and yellowish-colored spots with tortuous microvessels on the surface were observed, and background coloration was clearly seen in the lesion. As endoscopic biopsy suggested a histologically high-grade dysplasia; the lesion was completely resected en bloc by endoscopic submucosal dissection (ESD). The resected specimen was confirmed to be a squamous cell carcinoma in situ with extensive foamy cells in the superficial mucosal layer. Immunohistochemically, the observed foamy cells were strongly positive for CD68, which is characteristic of xanthoma. The clinical course was favorable, and no recurrence was observed 2 years and 7 mo after ESD. CONCLUSION: Diffuse xanthoma concurrent with early esophageal cancer is extremely rare. The characteristic endoscopic features may assist endoscopists in diagnosing similar lesions.