罕见累及唾液腺的Rosai-Dorfman病1例报告及文献复习

doi:10.12439/kqhm.1005-4979.2026.05.010

《口腔颌面外科杂志》 ›› 2026, Vol. 36 ›› Issue (5): 425-429. doi: 10.12439/kqhm.1005-4979.2026.05.010

罕见累及唾液腺的Rosai-Dorfman病1例报告及文献复习

吕洋(), 秦蓉(), 员东星, 陈新华, 许少媛, 叶美红

河南科技大学附属黄河三门峡医院口腔科，三门峡 472000

收稿日期:2025-06-16 接受日期:2025-12-01 出版日期:2026-10-28 上线日期:2026-11-03
通讯作者: 秦蓉，副主任医师. E-mail：15139890811@163.com
作者简介:
吕洋，主治医师. E-mail：2358624866@qq.com

Rosai-Dorfman disease with rare involvement of the salivary glands: A case report and literature review

LÜ Yang(), QIN Rong(), YUN Dongxing, CHEN Xinhua, XU Shaoyuan, YE Meihong

Department of Stomatology, the Yellow River Sanmenxia Hospital Affiliated to Henan University of Science and Technology, Sanmenxia 472000, China

Received:2025-06-16 Accepted:2025-12-01 Published:2026-10-28 Online:2026-11-03

摘要/Abstract

摘要：

Rosai-Dorfman病（Rosai-Dorfman disease，RDD）又称淋巴窦组织细胞增生症伴巨淋巴结病，是一种病因和发病机制均不明确的良性增生性疾病，发病部位无特异性，罕见累及唾液腺，临床上易被误诊，目前治疗方法尚无统一定论。本文通过报道1例罕见累及唾液腺的RDD，并结合相关文献，对其临床表现、病因及治疗进行讨论，旨在提高口腔医生对RDD的认知及诊治水平。

关键词: Rosai-Dorfman病, 唾液腺肿瘤, 组织细胞增多症

Abstract:

Rosai-Dorfman disease (RDD), also known as sinus histiocytosis with massive lymphadenopathy, is a benign proliferative disorder of unknown etiology and pathogenesis. It can occur at non-specific anatomical sites, rarely involves the salivary glands, and is highly prone to misdiagnosis in clinical practice. Currently, no consensus has been reached regarding its treatment. This article reports a rare case of RDD involving the salivary glands and reviews the relevant literature to discuss its clinical manifestations, etiology, and treatment, aiming to improve oral clinicians' understanding, diagnosis, and management of this disease.

Key words: Rosai-Dorfman disease, salivary gland tumor, histiocytosis

中图分类号:

R782.7

吕洋, 秦蓉, 员东星, 陈新华, 许少媛, 叶美红. 罕见累及唾液腺的Rosai-Dorfman病1例报告及文献复习[J]. 《口腔颌面外科杂志》, 2026, 36(5): 425-429.

LÜ Yang, QIN Rong, YUN Dongxing, CHEN Xinhua, XU Shaoyuan, YE Meihong. Rosai-Dorfman disease with rare involvement of the salivary glands: A case report and literature review[J]. Journal of Oral and Maxillofacial Surgery(CN), 2026, 36(5): 425-429.

https://journal06.magtech.org.cn/Jweb_joms/CN/Y2026/V36/I5/425

图/表 5

图1 患者皮肤及局部肿物图片 A.胸前皮肤病变；B.颌面部皮肤病变。

Figure 1 Clinical photographs showing the patient's skin lesions and local mass

图2 患者入院后下颌下腺MRI图片 A、D.颜面部及颈部皮肤结节；B、E.左侧下颌下腺肿物；C、F.左侧腮腺结节。蓝色箭头所示为病灶。A—C为斜轴位T2加权成像，D—F为斜冠状位T2加权抑脂成像。

Figure 2 MRI images of the submandibular gland after admission

图3 切除后的肿物大体形态 A.肿物外观；B.肿物剖面呈灰黄色，质地坚实，有包膜。

Figure 3 Gross morphology of the resected mass

图4 左下颌下肿物和下颌下腺病理图片 A—B. HE染色图（×100）；C. HE染色图（×40）；D. S-100（+）（×100）；E. CD68（+）（×100）；F. CD1α（-）（×100）。

Figure 4 Histopathological images of the left submandibular mass and submandibular gland

表1 唾液腺常见肿瘤与RDD的鉴别诊断

Table 1 Differential diagnosis of common salivary gland tumors and RDD

项目	RDD		多形性腺瘤	腺淋巴瘤		黏液表皮样癌		腺样囊性癌
病理组织学特点	非朗格汉斯细胞组织细胞增生性病变，以大量淡染组织细胞增生为特征，可见典型“伸入现象”^［5］		由腺上皮及肌上皮细胞构成，伴黏液样或软骨样间质	肿瘤由上皮和淋巴样组织构成		由黏液细胞、表皮样细胞及中间细胞组成		主要由导管上皮和肌上皮样细胞组成
免疫组织化学特点	S-100（+）、CD68（+）、CD163（+）、CD1α（-） ^[4]		CK（+）、S-100（+）、GFAP（+） ^[6]	CK（+）、CEA（+）、S-100（部分+）^[6]		EMA（+）、CK5/6（部分+）、p63（部分+） ^[7]		CK7（+）、Calponin（+）、CD117（+）、Ki-67（+） ^[8]

参考文献 10

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