The median arcuate ligament syndrome (MALS) is characterized by abdominal pain, nausea, and vomiting attributed to compression of the celiac axis by a fibrous band (the median arcuate ligament) connecting the diaphragmatic crura. The pathophysiologic origin of these symptoms is not clearly understood. Theories invoking either a neurogenic or vascular origin for the clinical features associated with MALS have been proposed, but objective evidence to support these theories is lacking. We describe the clinical course and gastric myoelectrical features of a patient with postprandial epigastric pain, weight loss, gastroparesis, and gastric dysrhythmias in whom a diagnosis of MALS was established. Surgical decompression of the celiac axis in our patient resulted in resolution of abdominal pain, return to a full diet within 4 weeks without nausea or vomiting, improvement in radionuclide gastric emptying, and restoration of the gastric electrical rhythm to a normal 3 cycle/min conduction rate. This is the first demonstration of altered gastric myoelectrical activity in a patient with MALS. The regularization of the gastric electrical rhythm in our patient after surgical decompression of the celiac axis would support a neurogenic basis for the symptoms associated with MALS. MALS should be excluded in patients with idiopathic gastroparesis and unexplained epigastric pain.