Orolingual angioedema is an uncommon, albeit well-established, side effect of tissue plasminogen activator. Herein presented is an especially refractory case with multiple confounding factors which was successfully treated with a novel approach, thus circumventing impending partial glossectomy.

Authors’ active participation in multidisciplinary treatment plan of care and subsequent chart review regarding patient’s hospital stay in a neurocritical care unit from 3/2019-5/2019.

A 48-year-old African American man with a history of trisomy 21 (with baseline macroglossia and an oral fixation), hypertension, and diabetes mellitus presented to an outside hospital with acute onset right sided weakness, aphasia, and left gaze deviation. Patient was administered tPA prior to transfer to a comprehensive stroke center for further evaluation and treatment. By the time of arrival, he had developed severe orolingual angioedema requiring nasotracheal intubation prior to mechanical thrombectomy of his large vessel occlusion. Orolingual angioedema proved to be refractory to epinephrine, antihistamines, and steroid infusions. Additionally, repetitive trauma due to patient continuously biting his tongue resulted in persistent swelling after the initial period of stabilization, despite bite blocks and a trial of neuromuscular blockade with train of four monitoring. Patient required tracheostomy during this period as well. Conservative measures including onabotulinum toxin injections to the masseters and pterygoids and application of serial pressure dressings to the tongue ultimately resulted in reduction of orolingual edema to the point his tongue was able to again fit in his oral cavity without need for partial glossectomy.

A novel approach to refractory orolingual angioedema after alteplase administration is presented in which conservative measures including medication management and mechanical intervention with serial pressure dressings resulted in sufficient resolution of symptoms to avoid operative intervention.