Unusual Case of a Massive Macroglossia Secondary to Myxedema: A Case Report and Literature Review.

James C Melville, Kelsey D Menegotto, Timothy C Woernley, Blake D Maida, Ibrahim Alava

Journal: Journal of oral and maxillofacial surgery : official journal of the American Association of Oral and Maxillofacial Surgeons 2019;76(1):119-127

PMID: 28742994

Abstract

Macroglossia is classified as true macroglossia, which exhibits abnormal histology with clinical findings, and relative macroglossia, in which normal histology does not correlate with pathologic enlargement. This report describes an atypical case of morbidity with massive macroglossia secondary to myxedema; the macroglossia enlarged over a 3-month period before being presented to the Department of Oral and Maxillofacial Surgery, University of Texas Health Science Center at Houston (Houston, TX). Substantial enlargement of the tongue (16 cm long × 10 cm wide) was first attributed to angioedema, which was refractory to the discontinuation of lisinopril and a C1 esterase inhibitor. A core tongue biopsy examination was performed to rule out angioedema, amyloidosis, myxedema, and idiopathic muscular hypertrophy. Interstitial tissue was positive for Alcian blue and weakly positive for colloidal iron, which are correlated with hypothyroidism and a diagnosis of myxedema. However, the macroglossia did not resolve after correcting for hypothyroidism. The patient required a wedge glossectomy for definitive treatment. She recovered unremarkably, with excellent cosmesis and preservation of lingual and hypoglossal function. There are some case reports of massive macroglossia but none with myxedema as the primary etiology.

Copyright © 2017 American Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.

Address: Assistant Professor, Oral and Maxillofacial Surgery, Oral, Head and Neck Oncology and Microvascular Reconstructive Surgery, School of Dentistry, University of Texas Health Science Center at Houston, Houston, TX. Electronic address: [email protected].; Resident PGY-3, Oral and Maxillofacial Surgery, School of Dentistry, University of Texas Health Science Center at Houston, Houston, TX.; Chief Resident PGY-6, Oral and Maxillofacial Surgery, School of Dentistry, University of Texas Health Science Center at Houston, Houston, TX.; Assistant Professor, Otolaryngology, McGovern Medical School, University of Texas Health Science Center at Houston, Houston, TX.
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