Macroglossia secondary to prolonged prone positioning for management of COVID-19 respiratory failure

Background

Macroglossia or enlarged tongue1 is a rare and potentially life-threatening2 condition clinically defined as tongue protrusion beyond the teeth or alveolar ridge at rest.3 4 It may lead to complications such as pain, chronic tongue drying, superimposed infection, impaired speech, swallow dysfunction and even airway obstruction.5 Etiologies include endocrinological, metabolic or neoplastic infiltration; infectious or inflammatory conditions; trauma; and, rarely, prolonged prone positioning.6 There are currently no standard guidelines for the management of acute macroglossia. Here, we report the case of a 59-year-old woman who developed acute macroglossia following prolonged prone positioning for management of COVID-19.

Case presentation

A 59-year-old woman with obesity, type 2 diabetes mellitus, obstructive sleep apnoea and history of thyroid adenocarcinoma status post thyroidectomy with surgical-induced hypothyroidism was hospitalised for COVID-19-induced respiratory failure. Acute care management included convalescent plasma, tocilizumab, intubation and mechanical ventilation for over 33 days with prone positioning for a total of 11 days (table 1). Macroglossia was noted on day 8 (table 1) of prone positioning, and a bedside tracheostomy was subsequently placed to bypass the oropharynx. Macroglossia was treated with Biotene gauze overlaid by Coban dressing and a Decadron taper. Prior to discharge from acute care, thyroid hormone assays were obtained revealing an elevated thyroid-stimulating hormone of 8.15 mIU/L in the setting of a normal free thyroxine level of 0.9 ng/dL. These findings suggested recovery from non-thyroidal illness in the absence of untreated hypothyroidism.

Following her 42-day acute care course, she was transferred to our facility for intensive inpatient rehabilitation. At this time, Coban dressing was discontinued due to intolerance. On arrival, she was unable to speak due to severe macroglossia, with her tongue protruding 10 cm beyond the oral cavity, 6 cm wide and 4 cm thick (figure 1). The tongue was encased in a keratinised plaque of dried-up necrotic tissue (figures 1 and 2). Recommendations from ear, nose and throat doctor were to use bite blocks (figures 3 and 4) to facilitate lymphatic and venous drainage or to have the patient undergo a partial glossectomy. The smallest bite guard (figures 3 and 4) available was introduced but subsequently removed due to intolerance for more than 5 min.

Despite debridement, keratinised plaque typically reaccumulated (figure 5). Dressings consisting of a gauze soaked in a mixture of Biotene and glycerin were placed around her tongue and covered with tubular elastic dressing for graded, even compression (figure 6). The dressings were changed twice a day. The tubular elastic dressing was suspended against gravity by two strings in order to mitigate vascular congestion by facilitating forward drainage through the normal venous and lymphatic channels (figures 6 and 7). Dressing changes with Biotene and glycerin-soaked gauze overlaid by suspended tubular elastic dressing were continued twice daily, allowing her to exercise her tongue for 30–45 min prior to each dressing change. The overall goal was to keep the tongue moist and elevated while applying even compression.

Outcome and follow-up

By day 8 of her acute rehab stay, the size of her tongue was significantly reduced; keratinised tissue was completely softened and easily removed, exposing normal-appearing tongue surface (figure 8). By day 10, her tongue was clean with the exception of a small amount of debris, which was cleaned with ease. At the time of discharge, on day 14, she was able to move her tongue freely and to intermittently retract it within the oral cavity (figure 9). By day 3 postdischarge, she was able to keep her tongue inside her oral cavity and communicate normally (figure 10).