Recurrent macroglossia requiring tracheostomy after haemorrhagic basal ganglia stroke

BMJ Case Rep. 2021 Jan 11;14(1):e238775. doi: 10.1136/bcr-2020-238775.

Abstract

A 50-year-old African American woman with hypertension, congestive heart failure, chronic kidney disease and prior cerebral vascular accident was transferred from an outside hospital after being found unresponsive and subsequently intubated for severe orolingual swelling. Imaging showed left thalamic haemorrhagic stroke, and the lingual swelling was clinically concerning for angio-oedema, with which a lingual biopsy was consistent. Work-up was negative for hereditary or acquired angio-oedema, and imaging was negative for structural causes. Of note, the patient had an episode of severe orolingual swelling 3 months prior to this presentation after suffering left thalamic haemorrhage which self-resolved after approximately 2 months. In both episodes lingual swelling predated receipt of tissue plasminogen activator and she had discontinued ACE inhibitor therapy since her first episode of tongue swelling. Despite medical and supportive management, tongue swelling progressed during admission and the decision was made to allow the patient's tongue swelling to self-resolve.

Keywords: dentistry and oral medicine; head and neck surgery; oral and maxillofacial surgery; otolaryngology / ENT; stroke.

Publication types

  • Case Reports

MeSH terms

  • Basal Ganglia Diseases / complications*
  • Basal Ganglia Diseases / diagnosis
  • Basal Ganglia Diseases / therapy
  • Female
  • Hemorrhagic Stroke / complications*
  • Hemorrhagic Stroke / diagnosis
  • Hemorrhagic Stroke / therapy
  • Humans
  • Macroglossia / diagnostic imaging
  • Macroglossia / etiology*
  • Macroglossia / therapy*
  • Middle Aged
  • Recurrence
  • Tracheostomy*