TRAUMATIC MACROGLOSSIA: A CASE REPORT

Abstract

TOPIC: Critical Care TYPE: Fellow Case Reports INTRODUCTION: Self-inflicted traumatic macroglossia is extremely rare and only reported in a handful of case reports. We re-port an interesting case in a 37-years-old female. CASE PRESENTATION: A 37-years-old female was admitted to ICU for intracranial hemorrhage. Hospital stay was complicated by acute respiratory failure requiring urgent intubation. On day 3 of admission, patient developed acute tongue swelling. Physical exam was remarkable for markedly enlarged tongue measuring 'length 10cm, width 9.5cm, and circumference 23 cm. An initial diagnosis of angioedema was made and the patient received multiple dos-es of steroids, antihistamine and even icatibant trial with minimal improvement. C1 esterase inhibitor, C1 es-terase antigen, C3, C4 and tryptase level were all unremarkable. Factor XII was also normal. Paraneoplastic workup including CT neck/chest/abdomen/pelvis and lumbar puncture were unremarkable. Tongue biopsy re-vealed edematous benign squamous mucosa negative for vasculitis. During her hospital course, she was wit-nessed actively biting her tongue -despite using a bite blocker- while she was awake for which she underwent tooth extraction (teeth #23–25) and had a jaw spreader placed. DISCUSSION: Traumatic macroglossia has been reported in literature following oral cavity surgeries, trauma in anticoagulated patients and tongue biting in epileptics etc. Self-inflicted traumatic macroglossia is rare but can be potentially life-threatening if airway is not secured promptly. Our patient was intubated before she developed tongue swelling. We initially treated her for angioedema given sudden onset but persistent tongue swelling for more than 30 days despite receiving multiple medications made the diagnosis of angioedema unlikely. Factor XII genetic analysis is used to identify patients with hereditary angioedema with normal C1 Inhibitor. Both C1 inhibitor and Factor XII were normal in our patient. A detailed review of medications causing tongue swelling as possible side effect was also done and possible culprits were discontinued. The patient underwent extensive imaging studies that ruled out SVC syndrome or other vascular/lymphatic obstruction as a potential cause. A diagnosis of self-inflicted traumatic mac-roglossia was made after witnessing the patient bite her tongue repeatedly in light of negative workup for other etiologies Tongue swelling was managed with tooth extractions, jaw spreaders, compressive dressing and lin-gual massage. Surgery was discussed with family but they opted for conservative management. CONCLUSIONS: This case report brings into light an interesting case of self-inflicted traumatic macroglossia as well as clinical challenges in its diagnosis and management. REFERENCE #1: A. J. Hotaling and J. S. Reilly, "The diagnosis and treatment of macroglossia in children," Ear, Nose, & Throat Journal, vol. 65, no. 10, pp. 444–448, 1986 REFERENCE #2: J. E. Vogel, J. B. Mulliken, and L. B. Kaban, "Macroglossia," Plastic and Reconstructive Surgery, vol. 78, no. 6, pp. 715–723, 1986 REFERENCE #3: B. W. Neville, D. D. Damm, A. Allen, and A. Chi, Oral and Maxillofacial Pathology, Elsevier Health Sciences, Amsterdam, Netherlands, 2015 DISCLOSURES: No relevant relationships by Sarah Elsayed, source=Web Response No relevant relationships by Ahmed Gohar, source=Web Response no disclosure on file for Zinobia Khan; No relevant relationships by Sudhir Krishnan, source=Web Response No relevant relationships by Mani Latifi, source=Web Response No relevant relationships by Mariam Saeed, source=Web Response