Acute Macroglossia: The Conspicuous Brunt of Proning

ABSTRACT

Introduction: Macroglossia is defined as an enlargement of the tongue in the resting position, protruding beyond the teeth. Many cases have been reported secondary to patient positioning while under anesthesia, post oro-pharyngeal packing, trauma or surgery, and allergic or non-allergic angioedema. However, acute macroglossia in the ICU is a rare situation. In COVID-19 related severe ARDS, endotracheal intubation and prolonged proning is an important part of management of hypoxemic respiratory failure Prone positioning also has the potential to independently cause macroglossia. Awareness of this complication of proning could help mitigate morbidity in patients. Case report A 53-year-old diabetic, hypertensive overweight female with OSA presented with progressive shortness of breath and productive cough. She was mechanically ventilated in ICU in view of respiratory distress secondary to COVID-19 pneumonia. Intubation was minimally traumatic with minimal oral bleeding settling over few minutes. Though managed with medications as per COVID-19 management protocol, she progressed to severe ARDS hence proned on hospital day 1 for 16 hours - PaO2/FiO2 ratio improved. She was not proned further. Acute macroglossia (3 inches outside the oral cavity) with lower facial edema was noted 4 days post proning. Saline moistened gauze was loosely wrapped around the tongue every hour. Circumferential ecchymosis was noticed around her neck on the 6th day. On day 8, macroglossia did not show signs of resolution. Hence, the tongue was pushed in manually every 2 hourly and the position was maintained manually for 10 minutes. The swelling decreased gradually with the tongue staying in a retracted position on treatment day 2. On day 3, there was a complete resolution of the swelling. However, she had persistent swallowing difficulty causing difficulty in weaning from tracheostomy. MRI of neck and chest showed large pre-vertebral collection from the upper border of C2 inferiorly across the thoracic inlet to the posterior mediastinum with thin linear extension up to the lower border of T4. Mass effect with airway compression, displacement, and compression of esophagus and neck vessels was seen. Trans-oral exploration revealed mucosal rent and bulge in the posterior pharyngeal wall. The hematoma was evacuated by ENT specialists. She was discharged on tracheostomy and feeding tube. Tracheostomy decannulation was done after 1 month. Discussion: In our practice of proning patients with ARDS for >10 years, this is the first case of macroglossia as a complication of proning that we encountered. Other factors that could have contributed to this patient are obesity and mildly traumatic intubation. Development of macroglossia 4 days after proning and resolution over a short period of time is rare and suggests lymphatic and vascular compression as the cause. Later development of ecchymosis and dysphagia may be due to the posterior pharyngeal injury. Conclusion: Proning, especially in obese patients, can be a challenge. Positioning of the face and avoidance of injury to any of the structures is vital to the care of the proned patient. Medical staff involved in patient care should be aware and vigilant to pick up this condition early to avoid further injury.