Purpose: Eosinophilic esophagitis (EoE) is characterized by an eosinophilic infiltration of the esophagus and is increasingly recognized as a cause of dysphagia and heartburn unresponsive to antireflux therapy. We report a case of eosinophilic esophagitis that presented without dysphagia, heartburn or endoscopic abnormalities to alert clinicians to the importance doing of esophageal biopsies in atypical presentations of EoE. Methods: A 19 yo college student presented with a 3 year history of anorexia and dyspepsia. Multiple foods and milk seemed to cause dyspepsia. However, his symptoms persisted despite avoiding milk and milk products. He had several episodes of minor hematemesis that led to an endoscopy at age 16 (by a different physician) showing only a small Mallory-Weiss tear and normal duodenal biopsies. Because of worsening symptoms and weight loss of ten pounds, he was referred for further evaluation. Family history was notable for mother with celiac sprue. A gastric emptying test was normal, LFTs and CBC were normal except for borderline eosinophilia; WBC = 7000/uL and 7.3% eosinophils (normal 0–7.0%). A full panel of celiac antibodies were negative except for an antigiadin IgG of 100.0 U/ml (normal <10.0 U/ml). A repeat endoscopy to assess for celiac sprue was normal except for slight gastric retention. Multiple biopsies were negative for celiac sprue and H. pylori. There was no eosinophilia in the duodenal or gastric biopsies. The esophagus was endoscopically normal without evidence of esophagitis, rings, or any other abnormalities. Biopsies from a normal appearing GE junction revealed a focal dense eosinophilic infiltration in the squamous epithelium (≥28 eosinophils per high powered field) consistent with EoE. The patient was started on fluticasone swallowed twice a day with resolution of all symptoms within a few weeks. Results: EoE is becoming more frequently diagnosed as a cause of dysphagia and heartburn and is predominantly considered in younger patients presenting with symptoms that are unresponsive to antireflux therapy or with endoscopic signs of EoE such as ringed esophagus. Diagnosis is by clinical features supported by esophageal biopsy with ≥15 eosinophils per HPF. Five biopsies (including proximal esophagus) have a sensitivity of 100%. Other bloodwork and endoscopic findings are neither sensitive nor specific. Conclusion: This case highlights the importance of esophageal biopsies in the diagnosis of EoE in atypical presentations. We suggest that clinicians consider the possibility of EoE in patients with prolonged, non-specific upper GI complaints and biopsy the esophagus even if the mucosa appears normal on endoscopy.