Melville James C, Menegotto Kelsey D, Woernley Timothy C, Maida Blake D, Alava Ibrahim
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.
Resident PGY-3, Oral and Maxillofacial Surgery, School of Dentistry, University of Texas Health Science Center at Houston, Houston, TX.
J Oral Maxillofac Surg. 2018 Jan;76(1):119-127. doi: 10.1016/j.joms.2017.06.033. Epub 2017 Jun 30.
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.
巨舌症分为真性巨舌症（其组织学异常伴有临床症状）和相对性巨舌症（其正常组织学与病理性肿大不相关）。本报告描述了一例黏液性水肿继发大量巨舌症的非典型发病病例；在该病例中，巨舌症在3个月内逐渐肿大，之后被转诊至德克萨斯大学休斯顿健康科学中心口腔颌面外科（德克萨斯州休斯顿）。舌体显著肿大（长16厘米×宽10厘米）最初被归因于血管性水肿，停用赖诺普利和C1酯酶抑制剂均无效。进行了舌部核心活检以排除血管性水肿、淀粉样变性、黏液性水肿和特发性肌肉肥大。间质组织阿尔辛蓝染色阳性，胶体铁染色弱阳性，这与甲状腺功能减退及黏液性水肿的诊断相关。然而，纠正甲状腺功能减退后巨舌症并未消退。患者需要进行楔形舌切除术以进行确定性治疗。她恢复良好，外形美观，舌部和舌下神经功能得以保留。有一些关于大量巨舌症的病例报告，但没有以黏液性水肿为主要病因的。
