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上牙龈局限性淀粉样变性:一例报告

Localized amyloidosis of the upper gingiva: a case report.

作者信息

Bucci Tommaso, Bucci Eduardo, Rullan Ana Maria Puig, Bucci Paolo, Nuzzolo Paolo

机构信息

Universiy Federico II, Via S, Pansini, 5 80131 Naples, Italy.

出版信息

J Med Case Rep. 2014 Jun 17;8:198. doi: 10.1186/1752-1947-8-198.

Abstract

INTRODUCTION

Localized amyloidosis in the head and neck is a rare and generally benign condition. In the oral cavity, amyloidosis usually involves the tongue or buccal mucosa. We present the second case of oral amyloidosis arising in the gingiva ever reported, to the best of our knowledge.

CASE PRESENTATION

A 73-year-old White Spanish man presented a persistent nodular mass involving his upper gingiva. The lesion was surgically resected and the histological examination revealed a subepithelial, multinodular amorphous and fibrillar accumulation. Staining of the specimen for Congo red proved positive, exhibiting a reddish colour under light microscopy and apple-green birefringence under polarized light. With immunohistochemical tests, pentagonal amyloid component was demonstrated. An extensive study excluded any systemic involvement; a diagnosis of localized primary amyloidosis was made. After 2 years of follow-up, no clinical progression to systemic amyloidosis or local recurrence was observed.

CONCLUSIONS

Localized amyloidosis of the gingiva is an extremely rare condition that seems to show no clinically distinct feature. Histologic examination is the first step towards diagnosis, followed by immunohistochemical tests. The diagnosis of localized amyloidosis should always be integrated with blood tests, a bone marrow biopsy, echocardiography and digestive endoscopy to intercept systemic involvement.

摘要

引言

头颈部局限性淀粉样变性是一种罕见且通常为良性的病症。在口腔中,淀粉样变性通常累及舌头或颊黏膜。据我们所知,我们报告了第二例牙龈发生的口腔淀粉样变性病例。

病例报告

一名73岁的西班牙白人男性上牙龈出现一个持续性结节状肿物。该病变经手术切除,组织学检查显示上皮下多结节状无定形和纤维状积聚。标本刚果红染色呈阳性,在光学显微镜下呈红色,在偏振光下呈苹果绿双折射。免疫组织化学检测显示有五角形淀粉样成分。全面检查排除了任何系统性受累;诊断为局限性原发性淀粉样变性。经过2年随访,未观察到向系统性淀粉样变性的临床进展或局部复发。

结论

牙龈局限性淀粉样变性是一种极为罕见的病症,似乎没有明显的临床特征。组织学检查是诊断的第一步,随后进行免疫组织化学检测。局限性淀粉样变性的诊断应始终结合血液检查、骨髓活检、超声心动图和消化内镜检查以发现系统性受累情况。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/03d2/4077115/a545588ce000/1752-1947-8-198-1.jpg

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