Adamo Daniela, Gasparro Roberta, Marenzi Gaetano, Mascolo Massimo, Cervasio Mariarosaria, Cerciello Giuseppe, De Novellis Danilo, Mignogna Michele Davide
Clinical Assistant, Oral Medicine Unit, Department of Neuroscience, Reproductive Sciences and Dentistry, University of Naples Federico II, Naples, Italy.
Resident, Oral Surgery Unit, Department of Neuroscience, Reproductive Sciences and Dentistry, University of Naples Federico II, Naples, Italy.
J Oral Maxillofac Surg. 2020 Sep;78(9):1572-1582. doi: 10.1016/j.joms.2020.04.022. Epub 2020 Apr 27.
Localized amyloidosis of the tongue is a benign condition in which surgical management may be considered. The aim of the study was to review the current literature and report a case.
We searched the PubMed database for all relevant articles reporting cases of localized tongue amyloidosis published between 1980 and February 2020. In addition, we updated 1 case diagnosed and treated in our department.
A 49-year-old male patient presented with an asymptomatic tongue nodule of the dorsum mimicking median rhomboid glossitis. The results of an incisional biopsy showed an amyloid on Congo red staining and positive findings for the κ light chain by immunohistochemical analysis. The findings of the systemic workup were negative. Therefore, a diagnosis of localized κ light-chain amyloidosis was made. The patient underwent a resection of the lesion, and no recurrence or progression was observed during a period of 18 months. The literature review showed 12 reports describing 21 patients (11 men, 52.3%) with localized tongue amyloidosis. The most common clinical presentation was nodular with a single lesion of the tongue dorsum (15 patients, 71.4%). All cases showed positive findings on Congo red staining. Immunohistochemical analysis findings were available for only 9 patients (42.8%) and showed light-chain amyloidosis. No case showed any systemic involvement or the development of systemic disease. Surgical excision was performed in 9 cases, with recurrence at the site of operation in 2 cases.
Localized amyloidosis of the tongue is a rare disease in which surgical excision may be therapeutic when a multidisciplinary evaluation does not show any systemic disease. We recommend an excision when the lesion is persistent or shows an enlargement or when discomfort is reported. In the case of any further local recurrence, resection may be repeated.
舌部局限性淀粉样变性是一种可考虑手术治疗的良性疾病。本研究的目的是回顾当前文献并报告一例病例。
我们在PubMed数据库中检索了1980年至2020年2月期间发表的所有报告舌部局限性淀粉样变性病例的相关文章。此外,我们更新了1例在我科诊断和治疗的病例。
一名49岁男性患者出现舌背无症状结节,类似正中菱形舌炎。切开活检结果显示刚果红染色呈淀粉样,免疫组化分析κ轻链呈阳性。全身检查结果为阴性。因此,诊断为局限性κ轻链淀粉样变性。患者接受了病变切除,在18个月期间未观察到复发或进展。文献回顾显示有12篇报告描述了21例(11例男性,占52.3%)舌部局限性淀粉样变性患者。最常见的临床表现为结节状,舌背单个病变(15例患者,占71.4%)。所有病例刚果红染色均呈阳性。仅9例患者(占42.8%)有免疫组化分析结果,显示为轻链淀粉样变性。没有病例显示有任何全身受累或全身性疾病的发生。9例患者进行了手术切除,2例在手术部位复发。
舌部局限性淀粉样变性是一种罕见疾病,当多学科评估未显示任何全身性疾病时,手术切除可能具有治疗作用。我们建议在病变持续存在、增大或有不适症状时进行切除。如果出现任何进一步的局部复发,可重复切除。