成人下顎骨之蘭格罕氏組織細胞增多症－病例報告

蘭格罕氏細胞組織球增生症爲罕見的疾病，病因目前未明。本文報告一64歲婦女，其主訴爲右下顎區腫塊數月且合併輕度疼痛，影像學檢查發現於右側下顎骨前下緣有皮質骨穿孔，經相關檢查及手術治療後，確認爲發生於顎下間隙之蘭格罕氏細胞組織球增生症，因有多處病灶，故照會腫瘤科進行放射線及化學治療，口腔部分則定期追蹤並採取保守治療。由於鄰近區域牙齒並無明顯動搖度和症狀，僅針對鄰近區域牙齒進行如牙周及齒髓相關治療及追蹤。2007年至2009年追蹤期間，陸續於左上顎後牙區齒槽骨處及右下顎第一大臼齒牙根處發現繼發性病灶，而原發現之病灶則無明顯持續破壞情形。自首次發現病灶至今已逾四年，目前仍持續追蹤觀察中。本病例因其原發部位爲軟組織病灶侵犯至下顎骨下緣，並造成該部位皮質骨破壞，由於初期病灶不典型，加上病息年紀較長，相關病徵亦與牙周病等類似而不易區分，診斷較爲困難。特此提出此病例，藉以提高臨床醫師之警覺性。

關鍵字

蘭格罕氏細胞組織球增生症；下顎骨；成人

並列摘要

Langerhans cell histiocytosis (LCH) is a rare disease with unknown etiology. We present an adult patient who suffering from LCH with an atypical clinical manifestation in the mouth. A 64-year-old female complained of right mandibular mass with mild pain for months. Radiographic examinations revealed a cortical bone perforation at right mandibular border. After surgical intervention, the pathological examination confirmed the disease entity to be the Langerhans cell histiocytosis. Because multifocal lesions were found, oncologists were consulted for adjuvant radiotherapy and chemotherapy. As asymptomatic lesions presented in the mandible, periodic periodontal and endodontic treatment for intraoral lesions were performed. We have kept follow-up on this patient for 4 years since the primary lesion were noted. Although primary lesions remaining stationary, secondary lesions developed at upper left posterior and at lower right posterior teeth region between 2007 and 2009. The initial manifestation of LCH in this patient was a soft tissue lesion involved into lower border of mandible causing cortical bone destruction. LCH rarely occurs in the adult patients. The manifestations of LCH in the mouth might mimic the signs of periodontal diseases and lead to delay in diagnosis. We thus present this case report in order to remind clinicians to include the LCH in differential diagnosis of such osteolytic lesions of jaw bone.

並列關鍵字

Langerhans cell histiocytosis ； mandible ； adult

國際替代計量

成人下顎骨之蘭格罕氏組織細胞增多症－病例報告

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