一例病理性下颌骨骨折，显示朗格汉斯细胞组织细胞增多症的双灶性病变。
A pathologic mandibular fracture revealing a bifocal location of Langerhans cell histiocytosis.
作者信息
Sabrine Maamouri, Marouen Ben Rejeb, Riahi Ines, Karima Zitouni, Nadia Zanidi, Issam Zairi
机构信息
Department of Maxillo Facial and Aesthetic Surgery of Charles Nicole's Hospital, Tunis, Tunisia.
Department of ORL of Charles Nicole's Hospital, Tunis, Tunisia.
出版信息
Ann Med Surg (Lond). 2020 Jun 26;56:128-132. doi: 10.1016/j.amsu.2020.06.019. eCollection 2020 Aug.
INTRODUCTION
Langerhans cell histiocytosis is a rare disease. When it occurs in the cranium/facial bones, the mandibular location is the most frequent.
PRESENTATION OF CASE
A 31 years-old man was referred to our department for a mandibular chronic discomfort during an acute exacerbation, a pathologic mandibular fracture.The diagnosis was confirmed by a subsequent CT scan. Histological and immunohistochemistry analysis of the maxillary and mandibular cystic lesions Langerhans cell histiocytosis of the oral cavity.
DISCUSSION
This is a case of rare single system LCH at two distinct locations: one at the mandibular bone and the other at the upper left maxilla. Both were uncovered by an acute exacerbation of a chronic discomfort secondary to a mandibular pathologic fracture. This should draw attention to the issues of the diagnosis.
CONCLUSION
An early LCH diagnosis and a multidisciplinary treatment plan allow the improvement of the patient 's prognosis and quality of life.
引言
朗格汉斯细胞组织细胞增多症是一种罕见疾病。当它发生于颅骨/面骨时，下颌骨部位最为常见。
病例介绍
一名31岁男性在急性加重期因下颌慢性不适、病理性下颌骨骨折被转诊至我科。随后的CT扫描确诊了该病情。对上颌和下颌囊性病变进行组织学和免疫组化分析，诊断为口腔朗格汉斯细胞组织细胞增多症。
讨论
这是一例罕见的单系统朗格汉斯细胞组织细胞增多症，累及两个不同部位：一处在下颌骨，另一处在左上颌骨。二者均因下颌病理性骨折继发的慢性不适急性加重而被发现。这应引起对诊断问题的关注。
结论
早期诊断朗格汉斯细胞组织细胞增多症并制定多学科治疗方案可改善患者预后和生活质量。
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