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一名患有下颌骨病理性骨折及自发性骨不连的成年多灶性朗格汉斯细胞组织细胞增多症病例报告。

Multifocal Langerhans cell histiocytosis in an adult with a pathological fracture of the mandible and spontaneous malunion: A case report.

作者信息

Shi Sailang, Liu Yanming, Fu Tao, Li Xiuzhen, Zhao Shifang

机构信息

Dental Clinic, Sir Run Run Shaw Hospital, School of Medicine, Zhejiang University, Hangzhou, Zhejiang 310016, P.R. China.

Department of Oral and Maxillofacial Surgery, Second Affiliated Hospital, Zhejiang University School of Medicine, Hangzhou, Zhejiang 310009, P.R. China.

出版信息

Oncol Lett. 2014 Sep;8(3):1075-1079. doi: 10.3892/ol.2014.2272. Epub 2014 Jun 20.

Abstract

Langerhans cell histiocytosis (LCH) is rare in the adult population and even rarer with jaw involvement. The current study presents the case of a 39-year-old male who complained of recurrent pain, swelling of the gingiva and an occasional pus-like discharge in the right mandible for one year. The patient was previously prescribed antibiotics, but this did not resolve the problem. An initial panoramic radiograph showed an osteolytic lesion and bone fracture in the right mandible. Eight months later, a new radiograph showed the spontaneous malunion of the fractured mandible. The patient was eventually diagnosed with Langerhans cell histiocytosis by histopathology and immunohistochemistry. Further lesions were found in the ribs and ilium by nuclear bone scanning. The patient was subsequently treated with systemic chemotherapy, and the lesions are currently effectively being controlled. This study is the first to show that spontaneous intralesional bone regeneration may lead to reunification of the mandible fracture caused by LCH in an adult.

摘要

朗格汉斯细胞组织细胞增多症(LCH)在成人中罕见,累及颌骨者更为罕见。本研究报告了一例39岁男性病例,该患者主诉右侧下颌骨反复疼痛、牙龈肿胀且偶尔有脓性分泌物,症状持续一年。患者此前曾服用抗生素,但问题未得到解决。最初的全景X线片显示右侧下颌骨有溶骨性病变和骨折。八个月后,新的X线片显示骨折的下颌骨自行愈合。患者最终通过组织病理学和免疫组织化学诊断为朗格汉斯细胞组织细胞增多症。核素骨扫描在肋骨和髂骨发现了其他病变。患者随后接受了全身化疗,目前病变得到有效控制。本研究首次表明,病灶内骨的自发再生可能导致成人LCH所致下颌骨骨折的愈合。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/eb9d/4114718/f4ee1431ba51/OL-08-03-1075-g00.jpg

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