MACROGLOSSIA IN THE SETTING OF COVID-19

SESSION TITLE: COVID-19 Case Report Posters 1 SESSION TYPE: Case Report Posters PRESENTED ON: 10/17/2022 12:15 pm - 01:15 pm INTRODUCTION: Macroglossia is a rare but life-threatening symptom that disrupts a person's ability to talk, swallow, and can also compromise their airway. Although not very well studied, there are several case reports describing a possible association between COVID-19 infection and macroglossia in people with African ancestry. We present an African American man who developed significant macroglossia several days after testing positive for COVID-19. CASE PRESENTATION: A 59 y/o African American male with a history of chronic bronchitis and tobacco use presented with 4 days of dyspnea. Sars-Cov-2 PCR was positive. Chest x-ray revealed bilateral, diffuse lung infiltrates. He had an elevated CRP of 295 and a d-dimer of 265. He became lethargic and hypercapnic requiring intubation which was nontraumatic. He was sedated, paralyzed, and proned. He received steroid therapy, broad spectrum antibiotics and a dose of Sarilumab. About a week later, he developed macroglossia that worsened over the course of days. Side effect profiles of each of his medications did not reveal any increased likelihood of macroglossia. C1Q complement cascade was mildly elevated and C1 esterase inhibitor level was normal. Diagnosis and treatment was necessary at this point as concerns for tongue central necrosis were raised and baseline tongue size would be required for proper evaluation and surgical intervention if necessary. He was given 4 units of FFP for possible angioedema with no improvement. CT Neck W/ contrast revealed edema and protrusion of the tongue without a discrete mass. Workup for acromegaly, sarcoidosis, amyloidosis, and hypothyroidism were negative. A pressure ulcer developed on his tongue due to the endotracheal tube and so he underwent tracheostomy. His tongue was draped in Chlorhexidine soaked gauze as well as Vashe wound solution. As he recovered from COVID-19 pneumonia, his respiratory status improved as well as his macroglossia. His tracheostomy was decannulated and his tongue returned to its baseline size. DISCUSSION: Macroglossia can lead to complications including airway compromise, dysphagia, or speech difficulties. It has been heavily proposed in the literature that COVID-19 infection can lead to postinfectious inflammatory peripheral nerve injury secondary to immune driven mechanisms. It was also previously proposed in literature based on immune-histochemical analysis of a tongue tissue sample taken from a COVID-19 patient that tongue muscle atrophy occurs as well as macrophage infiltration similar to that of nerve injury repair which can eventually lead to macroglossia. CONCLUSIONS: As the effects of COVID-19 are becoming better studied overtime, macroglossia, especially in those with African ancestry, is increasingly coming under the radar. This case report seeks to educate clinicians on this possible sequela and encourage supportive treatment in hopes that the tongue will recover. Reference #1: McCrossan S, Martin S, Hill C. Tongue Reduction for Macroglossia. J Craniofac Surg. 2021;32(5):1856-1859. doi:10.1097/SCS.0000000000007276 Reference #2: Colombo D, Del Nonno F, Nardacci R, Falasca L. May macroglossia in COVID-19 be related not only to angioedema?. J Infect Public Health. 2022;15(1):112-115. doi:10.1016/j.jiph.2021.10.026 Reference #3: Fernandez CE, Franz CK, Ko JH, et al. Imaging Review of Peripheral Nerve Injuries in Patients with COVID-19. Radiology. 2020;298 (3). https://doi.org/10.1148/radiol.2020203116 DISCLOSURES: No relevant relationships by Megan Devine No relevant relationships by Devin Haney No relevant relationships by Es-Haq Hassanin No relevant relationships by Nadim Islam No relevant relationships by Alyssa Weyer

Acute Macroglossia: The Conspicuous Brunt of Proning

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.

Traumatic Macroglossia in a Patient With Rett Syndrome.

Traumatic macroglossia is a rare condition characterized by a sudden edematous swelling of the tongue due to trauma that can progress into upper airway obstruction and asphyxia. We are presenting a case of a 20-year-old female with medical history significant for Rett syndrome who developed severe tongue swelling after multiple attempts of intubation secondary to low Glasgow Coma Scale (GCS) from a high dose of benzodiazepines. Traumatic macroglossia in this case was worsened further by uncontrolled bruxism. Multiple approaches were made to control the situation including placement of bite block, use of paralytics, and steroids. Multiple cases were reported about traumatic macroglossia but our case was unique in the sense that our patient did not respond well to conservative medical therapy and surgical approach was not possible as it was set to be done in a tertiary center; however, transferring the patient was not possible with the COVID-19 pandemic as hospitals were on diversion.

May macroglossia in COVID-19 be related not only to angioedema?

SARS-CoV-2 infection can lead to a variety of clinical manifestations. The occurrence of tongue swelling has recently reported in severe cases of COVID-19, and angioedema has suggested as the causative mechanism. Several factors, such as genetic predisposing factor and angiotensin-converting enzyme inhibitors (ACEI) therapies, have proposed to induce angioedema, especially as concerns patients requiring ICU treatments. Nevertheless, the question is still debated and other causes not yet recognized should be considered. Here we present a case of macroglossia occurred in a patient deceased for COVID-19 disease, who had no family history of angioedema and did not receive ACEI as antihypertensive drug. Histological and immune-histochemical analysis revealed tongue muscle atrophy with infiltrating macrophages suggesting repair mechanisms, as seen in nerve injury recovery. These new pathological findings may open new fields of study on the pathogenesis of SARS-CoV-2.

Are Oral Mucosal Changes a Sign of COVID-19? A Cross-Sectional Study at a Field Hospital. / ¿Son las alteraciones en la mucosa oral un signo de COVID-19? Estudio transversal en un Hospital de Campaña.

BACKGROUND: Coronavirus disease 19 (COVID-19) has many manifestations, including respiratory, thrombotic, neurologic, digestive, and cutaneous ones. Cutaneous manifestations have been classified into 5 clinical patterns: acro-ischemic (pseudo-chilblain), vesicular, urticarial, maculopapular, and livedoid. Oral manifestations have also been reported, but much less frequently. PATIENTS AND METHODS: We performed a cross-sectional study in which we examined the oral mucosa of 666 patients with COVID-19 at the IFEMA field hospital in Madrid in April 2020. RESULTS: Seventy-eight patients (11.7%) had changes involving the oral mucosa. The most common were transient anterior U-shaped lingual papillitis (11.5%) accompanied or not by tongue swelling (6.6%), aphthous stomatitis (6.9%), a burning sensation in the mouth (5.3%), mucositis (3.9%), glossitis with patchy depapillation (3.9%), white tongue (1.6%), and enanthema (0.5%). Most of the patients also reported taste disturbances. CONCLUSION: COVID-19 also manifests in the oral cavity. The most common manifestations are transient U-shaped lingual papillitis, glossitis with patchy depapillation, and burning mouth syndrome. Mucositis with or without aphthous ulcers or enanthema may also be observed. Any these findings may be key clues to a diagnosis of COVID-19.

Oral Complications of ICU Patients with COVID-19: Case-Series and Review of Two Hundred Ten Cases.

BACKGROUND: The critically ill patients suffering from coronavirus disease (COVID-19) and admitted to the intensive care units (ICUs) are susceptible to a wide array of complications that can be life-threatening or impose them to long-term complications. The COVID-19 oral mucocutaneous complications require multidisciplinary management and research for their pathophysiological course and epidemiological significance; therefore, the objective of this study was to evaluate the prevalence and characteristics of the critically ill COVID-19 patients with oral complications. METHODS: We described the clinical and microbiological characteristics of the critically ill COVID-19 patients in our ICU department (Banska Bystrica, Slovakia). In addition, we reviewed the current body of evidence in Ovid MEDLINE®, Embase, Cochrane Library, and Google Scholar for the oral mucocutaneous complications of ICU patients with COVID-19. RESULTS: Three out of nine critically ill patients (33.3%) in our ICU department presented with oral complications including haemorrhagic ulcers and necrotic ulcers affecting the lips and tongue. The microbiological assessment revealed the presence of opportunistic pathogens, confirming the possibility of co-infection. On reviewing the current literature, two hundred ten critically ill patients were reported to have oral complications due to their stay in the ICU setting. Perioral pressure ulcers were the most common complication, followed by oral candidiasis, herpetic and haemorrhagic ulcers, and acute onset macroglossia. The prolonged prone positioning and mechanical ventilation devices were the primary risk factors for those oral complications, in addition to the immunosuppressive drugs. CONCLUSIONS: The multidisciplinary approach is strongly advocated for monitoring and management of COVID-19 patients, thus implying that dermatology and oral healthcare specialists and nurses should be integrated within the ICU teams.