Consecutive Severe Orofacial Complications in Intensive Care Unit Patients: Quincke's Disease and Macroglossia due to Prolonged Prone Positioning for Management of Acute Respiratory Distress Syndrome

Nabil Belfeki; Souheil Zayet; Oumar Sy; Louis Marie Coupry; Sandy Mazerand; Ibrahim Chouchane; Cyrus Moini; Mehran Monchi; Arsène Mekinian

doi:10.12890/2022_003421

Consecutive Severe Orofacial Complications in Intensive Care Unit Patients: Quincke's Disease and Macroglossia due to Prolonged Prone Positioning for Management of Acute Respiratory Distress Syndrome

Eur J Case Rep Intern Med. 2022 Aug 24;9(8):003421. doi: 10.12890/2022_003421. eCollection 2022.

Authors

Nabil Belfeki¹, Souheil Zayet², Oumar Sy³, Louis Marie Coupry³, Sandy Mazerand³, Ibrahim Chouchane⁴, Cyrus Moini⁵, Mehran Monchi³, Arsène Mekinian⁶

Affiliations

¹ Department of Internal Medicine, Groupe Hospitalier Sud Ile de France, Melun, France.
² Department of Infectious Diseases, Hôpital Nord Franche-Comté, Trévenans, France.
³ Department of Intensive Medicine, Groupe Hospitalier Sud Ile de France, Melun, France.
⁴ Department of Imaging, Groupe Hospitalier Sud Ile de France, Melun, France.
⁵ Department of Cardiology, Groupe Hospitalier Sud Ile de France, Melun, France.
⁶ Department of Internal Medicine, Hôpital Saint Antoine, Assistante Publique des Hôpitaux de Paris (AP-HP), Paris, France.

Abstract

Critically ill patients admitted into the intensive care units are susceptible to a wide array of complications that can be life-threatening, or lead to long-term complications. Some complications are inherent to the patient's condition and others are related to therapeutics or care procedure. The prolonged prone positioning and mechanical ventilation devices are the first risk factors for orofacial complications. We report the case of a 47-year-old male patient, with a history of sleep apnoea syndrome, morbid obesity (body mass index of 43 kg/m2), and gastroesophageal reflux disease, presented to the emergency department with recent otorhinolaryngological symptoms of dysphonia and exertional dyspnoea lasting two days, and complicated with Quincke's disease. First-line treatment consisted of a compilation of intravenous antihistamines and corticosteroids. The patient's condition worsened. He developed an acute respiratory distress syndrome secondary to ventilator-acquired pneumonia with prone positioning ventilation, complicated by severe macroglossia. Soaked gauze dressings were placed around his tongue. Progressively, the size of his tongue reduced.

Learning points: Intensive care unit (ICU) patients are susceptible to a wide array of life-threatening complications that can be linked.Oral severe acquired Quincke's disease is an isolated form of angioneurotic oedema that is induced by several factors including gastroesophageal reflux disease, sleep apnoea, inhalation exposure, or drug reactions. Macroglossia is rare life-threatening complication due to prolonged prone positioning of unknown pathogenesis. An experienced critical care staff with standardised protocol is needed to prevent such a complication.Because of possible consecutive severe orofacial complications, prolonged prone positioning for management of acute respiratory distress syndrome (ARDS) is not recommended in patients with inaugural oral angioedema.

Keywords: Intensive care unit; Quincke’s disease; acute respiratory distress syndrome; macroglossia; prone positioning.