Duodenal stricture is a rare manifestation of celiac disease. In this case report, we present a case of a 64-year-old male with a history of duodenal stricture proven on both endoscopy and imaging, initially not responsive to endoscopic dilation. Further investigation and biopsy confirmed the diagnosis of celiac disease. In addition to endoscopic treatment, a gluten-free diet resulted in clinical, endoscopic, and histologic improvement. This case highlights the importance of considering celiac disease in the differential diagnosis for patients with duodenal strictures.