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颞下颌关节紊乱症揭示的朗格汉斯细胞组织细胞增多症:一例报告及颅面部表现综述

[Langerhans cell histiocytosis revealed by a temporomandibular joint disorder: Report of a case and review of the craniofacial expressions].

作者信息

Desbarats C, Adnot J, Bastien A V, Trost O

机构信息

Service de chirurgie maxillo-faciale et stomatologie, centre hospitalo-universitaire de Rouen, 1, rue de Germont, 76000 Rouen, France.

Service de chirurgie maxillo-faciale et stomatologie, centre hospitalo-universitaire de Rouen, 1, rue de Germont, 76000 Rouen, France; Laboratoire d'anatomie, faculté de médecine de Rouen, 22, boulevard Gambetta, 76000 Rouen, France; Institut national de la santé et de la recherche médicale (Inserm), LIMICS UMR-1142, 76000 Rouen, France.

出版信息

Rev Med Interne. 2020 Jan;41(1):50-53. doi: 10.1016/j.revmed.2019.08.005. Epub 2019 Aug 29.

Abstract

INTRODUCTION

Langerhans cell histiocytosis (HL) is a rare disease that can affect all tissues. Oral manifestations such as mucosal ulcer and tooth mobility are often the first signs of the disease. We report a rare case of mandibular condyle unifocal HL mimicking a temporomandibular joint disorder.

CASE REPORT

A 44-year-old patient presented with a left temporomandibular disorder with painful left preauricular swelling. The imaging assessment found a bone lesion of the left mandibular condyle. A curettage with biopsy was used to diagnose HL. Six months later, the patient had no more pain.

DISCUSSION

The craniofacial clinical expressions of HL mainly concern the bones, which can cause: pain, swelling, fracture, compression of noble organs. The other sites are: oral cavity, skin, lymph nodes, or eyes. Isolated forms are generally benign, and their treatment is discussed between abstention and non-aggressive surgery.

摘要

引言

朗格汉斯细胞组织细胞增多症(HL)是一种可累及所有组织的罕见疾病。口腔表现如黏膜溃疡和牙齿松动往往是该疾病的首发症状。我们报告一例罕见的下颌髁突单灶性HL,表现类似颞下颌关节紊乱病。

病例报告

一名44岁患者出现左侧颞下颌关节紊乱,伴有左侧耳前疼痛性肿胀。影像学评估发现左侧下颌髁突有骨质病变。通过刮除术加活检诊断为HL。6个月后,患者不再疼痛。

讨论

HL的颅面临床表现主要涉及骨骼,可导致:疼痛、肿胀、骨折、重要器官受压。其他部位包括:口腔、皮肤、淋巴结或眼睛。孤立型通常为良性,其治疗方案在观察等待和非侵袭性手术之间进行讨论。

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