Abstract

INTRODUCTION: Zollinger-Ellison Syndrome (ZES) is characterized by hypersecretion of gastrin from duodenal or pancreatic gastrinomas. Ectopic secretion of gastrin results in high gastric acid output due to the trophic action of gastrin on parietal cells and histamine-secreting ECL cells. ZES typically presents with symptoms of severe acid-related peptic ulcer disease such as abdominal pain, diarrhea, heartburn, and weight loss as well as complications from acid hypersecretion such as GI bleeding, stricture, and perforation. We report a novel case of a 60-year-old female who presented with hypersalivation as her chief complaint. CASE DESCRIPTION/METHODS: A 60-year-old female with type 2 diabetes mellitus, gastroparesis, and GERD presented to her gastroenterologist with nausea, hypersalivation and a “sour taste” in her mouth over the past few months. She states that this sour taste is constantly in her mouth and throat but is worse in the evenings. Her symptoms are associated with belching but she denies any heartburn. Medications at that time included pantoprazole 40 mg daily and ranitidine 150 mg BID. She initially underwent EGD with findings of chronic gastritis. Gastric biopsies were consistent with mild chronic gastritis with foveolar hyperplasia and intestinal metaplasia and staining for H.pylori was negative. Serum fasting gastrin level was significantly elevated at 1,379 pg/mL (normal < 100 pg/mL). CT abdomen/pelvis revealed a 13 × 12 mm subtly enhancing soft tissue density between the pancreatic head and the lumen of the duodenum, with concern for neuroendocrine tumor. The patient is scheduled to undergo further evaluation of these CT scan findings with endoscopic ultrasound and fine needle aspiration. DISCUSSION: This case is an example of an atypical presentation of ZES leading to delayed diagnosis and treatment. Symptoms of hypersalivation have been described in GERD, however the literature does not describe any cases of hypersalivation as a presenting symptom in patients with ZES. It is thought that reflux of stomach acid into the pharynx may lead to an increase in saliva production in order to neutralize the excess acid. The rare nature of ZES can lower clinical suspicion for the disorder and ultimately result in a delay in diagnosis. This case provides a unique example of ZES that highlights the importance of increased awareness of this disorder in patients who present with chronic GI symptoms despite adequate therapy.

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