Dystonia-associated features of anaphylaxis, including tongue swelling, and chest and throat tightness, have been rarely reported with selective serotonin reuptake inhibitor (SSRI) use. The patient is a 44-year-old woman who presented with palpitations, diaphoresis, dyspnea, swelling of the lips and tongue, and fixed upward deviation of her right eye following inadvertent ingestion of 20 mg of escitalopram in addition to her usual 10-mg dose. She reported transient resolution of all symptoms after autoinjector aqueous epinephrine administration (0.3 mg), with recurrence of symptoms after 35 min. The patient presented with one prior episode of anaphylactic symptoms and dystonia. She also reported one episode with purely anaphylactic features of swelling of lips and tongue, difficulty breathing and syncope. This case represents a unique dose-dependent episode of escitalopram-associated oculogyric dystonia with anaphylactic features. The transient resolution of the associated features of dystonia with intramuscular epinephrine administration is unique and suggests a common pathophysiology of the dystonic and anaphylactic symptoms.