Combination Lingual Compression and Maxillomandibular Fixation for Persistent Acquired Macroglossia: A Case Report

Acquired macroglossia is an uncommon condition with etiologies including trauma, idiopathic (post-operative), angioedema, infectious/inflammatory conditions, and neoplasms.1 Though often self-limited, severe cases causing oropharyngeal obstruction can result in airway compromise. While most cases can be managed conservatively with steroids and compression therapy,2, 3 persistent cases pose challenges, with only a few case reports describing alternative management options. In this report, we present a case of acute edematous macroglossia secondary to bite trauma, treated with a novel combination of manual tongue massage and maxillomandibular fixation (MMF). A 40-year-old African American female with a history of hypertension, hyperlipidemia, alcohol use disorder, obesity, and multinodular goiter, was admitted following an occlusive cerebrovascular accident managed by mechanical thrombectomy. Her course was complicated by cerebral edema and hemorrhagic conversion on heparin, requiring a decompressive hemicraniectomy. She remained ventilator dependent and underwent tracheostomy and percutaneous endoscopic gastrostomy (PEG) tube placement. She was then transferred to our tertiary care academic institution, where we were consulted for macroglossia. Upon evaluation, there was glossoptosis with significant protrusion, which had reportedly been present for greater than 2 weeks (Figure 1A). Initial conservative management with steroids, tongue massage, lymphedema therapy, compressive dressings, and a bite block was unsuccessful (Figure 2A). Operative management was discussed and the patient and her family were consented for manual reduction and MMF. Intraoperatively, there was significant tongue protrusion with desiccation and ulceration secondary to dental trauma. The tongue was manually reduced with 30 minutes of lymphatic massage (Figure 2B), then placed into the oral cavity with complete mouth closure and normal occlusion (Figure 2C). MMF was applied using the Synthes MatrixWAVE system (Warsaw, IN); interdental fixation was completed with a combination of wires and rubber bands (Figure 2D). At two weeks, the patient reported discomfort and intermittent bleeding from the MMF wires. The remaining MMF wires were removed and replaced with rubber bands. After four weeks, the patient returned to the operating room for hardware removal and debridement of small (0.5 × 1 cm) posterolateral tongue fibroma related to prior bite trauma. One week after MMF removal, the patient reported well-controlled pain, hemostasis, and continued improvement in edema (Figure 1B). With persistent therapy, she regained volitional control of tongue movement and was able to tolerate oral intake, enabling PEG tube removal. She was later decannulated without issue, and follow-up at 6 months demonstrated no macroglossia recurrence. Acute acquired macroglossia is uncommon problem with a variety of possible etiologies. Severe cases may result in airway compromise. Our patient had a tracheostomy in place prior to macroglossia development, thus there was no concern for respiratory compromise. In this case, glossal edema was attributed to bite injury and impaired lymphatic drainage from the anterior tongue secondary to compression by the teeth. Reduction glossectomy is a more invasive management option for macroglossia that can be indicated for true congenital, metabolic, or vascular etiologies.4 While generally effective, reduction glossectomy is a morbid procedure with moderate risk of dehiscence (13.5%), infection (1.35%), and recurrence (17.6%).5 Conversely, acquired macroglossia secondary to trauma is often managed conservatively with intravenous steroids, bite blocks, and massage with compression therapy. Anderson et al. and Johnson et al describe successful lingual compression therapy—one in the setting of angioedema with bite trauma, and one in a patient with COVID-19 requiring prolonged proning. Both used saline-moistened gauze and compressive wraps for 4 to 11 days, followed by manual reduction. These techniques, however, were inadequate in our case given the severity of our patient's glossoptosis. MMF has been previously described by Shanti et al. in a case of mild macroglossia following a gunshot wound to the tongue.6 However, our case differs in both mechanism and severity: their case involved minimal acute macroglossia due to penetrating ballistic trauma, while ours involved chronic non-penetrating bite trauma with significant glossoptosis and presumed lymphatic outflow obstruction. Given persistent macroglossia despite conservative measures as described above, our patient was taken to the operating room for a novel combination of manual tongue reduction and MMF with successful resolution. Despite the severity of her glossoptosis, this novel multimodal approach—conservative management, intraoperative lymphatic massage, and MMF—allowed rapid functional recovery with complete restoration of tongue function, decannulation, and resumption of oral feeding, while avoiding the morbidity and associated complications of a partial glossectomy. This study was approved by the Thomas Jefferson University Institutional Review Board and deemed IRB exempt. Emma De Ravin: clinical care, study design, manuscript drafting, manuscript revision, and submission; Annie Moroco: clinical care, study design, manuscript revision; Adam J. Luginbuhl: clinical care, study design, manuscript revision, and project oversight. None. None.