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原发性结外罗道尔夫-多夫曼病的颞骨表现:一例报告。

Temporal bone manifestation of primary extranodal Rosai-Dorfman disease: a case report.

机构信息

Division of Otolaryngology-Head and Neck Surgery, Department of Surgery, Cumming School of Medicine, University of Calgary, Calgary, Canada.

Department of Pathology and Laboratory Medicine, Arnie Charbonneau Cancer Institute, University of Calgary, Calgary, Canada.

出版信息

J Med Case Rep. 2023 Jun 21;17(1):280. doi: 10.1186/s13256-023-03790-8.

Abstract

BACKGROUND

Rosai-Dorfman disease is a rare benign histiocytic disorder characterized in most cases by painless cervical adenopathy. Less than 10% of extranodal cases involve bony lesions. Primary bone Rosai-Dorfman disease in the absence of nodal disease is extremely rare.

CASE PRESENTATION

A 48 year-old Caucasian male presented with progressive right-sided otalgia, tinnitus, vertigo, and hearing loss. A right temporal bone lytic lesion was detected on diagnostic imaging. Resection of the lesion and histopathological examination revealed Rosai-Dorfman disease.

CONCLUSIONS

Rosai-Dorfman disease primary bone lesions are an atypical presentation of a rare disease. This is the second reported case of Rosai-Dorfman disease arising within the temporal bone. This case study reveals that Rosai-Dorfman disease should be considered for patients presenting with inflammatory/lytic lesions of the temporal bone, in cases where infection and malignancy have been excluded.

摘要

背景

罗道氏病(Rosai-Dorfman disease)是一种罕见的良性组织细胞增生症,在大多数情况下表现为无痛性颈淋巴结病。不到 10%的结外病例涉及骨病变。在没有淋巴结疾病的情况下,原发性骨罗道氏病极为罕见。

病例介绍

一名 48 岁白人男性因右侧耳痛、耳鸣、眩晕和听力损失而就诊。诊断影像学显示右侧颞骨溶骨性病变。病变切除和组织病理学检查显示罗道氏病。

结论

罗道氏病原发性骨病变是一种罕见疾病的非典型表现。这是颞骨内罗道氏病的第二例报告病例。本病例研究表明,对于排除感染和恶性肿瘤的颞骨炎症/溶骨性病变患者,应考虑罗道氏病。

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