成年男性累及多处骨的朗格汉斯细胞组织细胞增生症1例

doi:10.3969/j.issn.1005-4979.2022.03.012

《口腔颌面外科杂志》 ›› 2022, Vol. 32 ›› Issue (3): 199-202. doi: 10.3969/j.issn.1005-4979.2022.03.012

• 病例报告 • 上一篇

成年男性累及多处骨的朗格汉斯细胞组织细胞增生症1例

刘洋(), 李龙江()

口腔疾病研究国家重点实验室，四川大学华西口腔医院头颈肿瘤外科，四川成都 610041

收稿日期:2021-03-12 修回日期:2021-08-26 出版日期:2022-06-28 发布日期:2022-06-29
通讯作者: 李龙江，教授. E-mail: muzili63@163.com.
作者简介:
刘洋（1994—），男，河南人，硕士研究生. E-mail: 595444525@qq.com

Langerhans cell histiocytosis involving multiple bones in an adult male: A case report

LIU Yang(), LI Longjiang()

State Key Laboratory of Oral Diseases, Department of Head and Neck Oncology, West China Hospital of Stomatology, Sichuan University, Chengdu 610041, Sichuan Province, China

Received:2021-03-12 Revised:2021-08-26 Online:2022-06-28 Published:2022-06-29

摘要/Abstract

摘要：

朗格汉斯细胞组织细胞增生症(Langerhans cell histiocytosis, LCH）是一种罕见的疾病，目前病因未明，病变可累及全身多部位。本文报道1例累及肋骨、髂骨及颌骨多处骨组织的LCH的成年男性患者，并结合相关文献对该疾病的流行病学特点、病因、临床特点、诊断、治疗和预后进行讨论。

关键词: 肋骨, 髂骨, 颌骨, 朗格汉斯细胞组织细胞增生症

Abstract:

Langerhans cell histiocytosis (LCH) is a rare disease. Its etiology is uncertain now and its lesions can involve multiple sites. In this study, a case that an adult male with LCH involving rib, ilium and jaw was presented, and the epidemiological characteristics, etiology, clinical characteristics, diagnosis, treatment and prognosis of this case were discussed in combination with relevant literatures.

Key words: rib, ilium, jaw, Langerhans cell histiocytosis

刘洋, 李龙江. 成年男性累及多处骨的朗格汉斯细胞组织细胞增生症1例[J]. 《口腔颌面外科杂志》, 2022, 32(3): 199-202.

LIU Yang, LI Longjiang. Langerhans cell histiocytosis involving multiple bones in an adult male: A case report[J]. 《Journal of Oral and Maxillofacial Surgery》, 2022, 32(3): 199-202.

https://journal06.magtech.org.cn/Jweb_joms/CN/Y2022/V32/I3/199

图/表 6

图1 髋关节CT示：左侧髋臼及邻近左侧髂骨多发不规则骨质破坏影

Figure 1 CT image showing multiple irregular bone destruction of left acetabulum and adjacent left ilium

图2 PET/CT示：左侧髋臼及邻近左侧髂骨处代谢增高

Figure 2 PET/CT image showing increased metabolism in the left acetabulum and adjacent left ilium

图3 术前曲面断层片示：右侧上颌骨及双侧下颌骨凹坑状吸收，边界尚清晰，边缘欠光滑

Figure 3 Tomography image showing the right maxilla and bilateral mandible were resorbed in pits, the boundary was still clear, and the edge was not smooth

图4 术中见左下颌骨磨牙区牙槽骨吸收，牙根暴露

Figure 4 Image of alveolar bone resorption and root exposure in the molar region of the left mandible during the operation

图5 术后病理苏木精-伊红（hematoxylin-eosin，HE）染色结果示：组织细胞增生并呈弥漫性浸润（×200）

Figure 5 Postoperative pathological hematoxylin-eosin (HE) staining showed that histiocytes proliferation and diffuse infiltration (×200)

图6 术后病理HE染色结果示：增生的组织细胞细胞核体积大，部分可见核分裂，部分细胞内可见空泡性变（×400）

Figure 6 Postoperative pathological HE staining showed that the nuclei of the proliferative tissue cells were large, some mitoses were seen, and some cells had vacuolar degeneration (×400)

参考文献 15

[1]	Zinn DJ, Chakraborty R, Allen CE. Langerhans cell his-tiocytosis: Emerging insights and clinical implications[J]. Oncology (Williston Park), 2016, 30(2): 122-132, 139.
[2]	Salotti JA, Nanduri V, Pearce MS, et al. Incidence and clinical features of Langerhans cell histiocytosis in the UK and Ireland[J]. Arch Dis Child, 2009, 94(5): 376-380. doi: 10.1136/adc.2008.144527 pmid: 19060008
[3]	Kobayashi M, Tojo A. Langerhans cell histiocytosis in adults:Advances in pathophysiology and treatment[J]. Cancer Sci, 2018, 109(12): 3707-3713. doi: 10.1111/cas.2018.109.issue-12 URL
[4]	Badalian-Very G, Vergilio JA, Degar BA, et al. Recurrent BRAF mutations in Langerhans cell histiocytosis[J]. Blood, 2010, 116(11): 1919-1923. doi: 10.1182/blood-2010-04-279083 pmid: 20519626
[5]	Allen CE, Merad M, McClain KL. Langerhans-cell histiocytosis[J]. N Engl J Med, 2018, 379(9): 856-868. doi: 10.1056/NEJMra1607548 URL
[6]	Krooks J, Minkov M, Weatherall AG. Langerhans cell histiocytosis in children: History, classification, pathobiology, clinical manifestations, and prognosis[J]. J Am Acad Dermatol, 2018, 78(6): 1035-1044. doi: S0190-9622(17)31930-8 pmid: 29754885
[7]	Erker C, Harker-Murray P, Talano JA. Usual and unusual manifestations of familial hemophagocytic lymphohistiocytosis and Langerhans cell histiocytosis[J]. Pediatr Clin North Am, 2017, 64(1): 91-109. doi: 10.1016/j.pcl.2016.08.006 URL
[8]	Haupt R, Minkov M, Astigarraga I, et al. Langerhans cell histiocytosis (LCH): Guidelines for diagnosis, clinical work-up, and treatment for patients till the age of 18 years[J]. Pediatr Blood Cancer, 2013, 60(2): 175-184. doi: 10.1002/pbc.v60.2 URL
[9]	Kilpatrick SE, Wenger DE, Gilchrist GS, et al. Langerhans′ cell histiocytosis (Histiocytosis X) of bone. A clinicopathologic analysis of 263 pediatric and adult cases[J]. Cancer, 1995, 76(12): 2471-2484. doi: 10.1002/1097-0142(19951215)76:12<2471::aid-cncr2820761211>3.0.co;2-z pmid: 8625073
[10]	Madrigal-Martínez-Pereda C, Guerrero-Rodríguez V, Guisado-Moya B, et al. Langerhans cell histiocytosis: Literature review and descriptive analysis of oral manifestations[J]. Med Oral Patol Oral Cir Bucal, 2009, 14(5): E222-E228.
[11]	Desbarats C, Adnot J, Bastien AV, et al. Langerhans cell histiocytosis revealed by a temporomandibular joint disorder: Report of a case and review of the craniofacial expressions[J]. Rev Med Interne, 2020, 41(1): 50-53. doi: S0248-8663(19)30622-8 pmid: 31474430
[12]	Darvishian F, Hirawat S, Teichberg S, et al. Langerhans' cell histiocytosis in the parotid gland[J]. Ann Clin Lab Sci, 2002, 32(2): 201-206. pmid: 12017204
[13]	Yang SD, Chen XM, Zhang JL, et al. Isolated Langerhans cell histiocytosis of the sublingual gland in an adult[J]. Int J Clin Exp Pathol, 2015, 8(10): 13647-13650.
[14]	DiCaprio MR, Roberts TT. Diagnosis and management of Langerhans cell histiocytosis[J]. J Am Acad Orthop Surg, 2014, 22(10): 643-652. doi: 10.5435/JAAOS-22-10-643 pmid: 25281259
[15]	Allen CE, Ladisch S, McClain KL. How I treat Langerhans cell histiocytosis[J]. Blood, 2015, 126(1): 26-35. doi: 10.1182/blood-2014-12-569301 pmid: 25827831

选择文件类型/文献管理软件名称

选择包含的内容

成年男性累及多处骨的朗格汉斯细胞组织细胞增生症1例

Langerhans cell histiocytosis involving multiple bones in an adult male: A case report

RichHTML

PDF

可视化

被引次数

摘要/Abstract

引用本文

使用本文

图/表 6

参考文献 15

相关文章 15

编辑推荐

Metrics

本文评价

[1]	胡娟. 游离组织瓣修复颌面部缺损后血管蒂骨化的发生率及影响因素的系统评价[J]. 《口腔颌面外科杂志》, 2023, 33(6): 379-384.
[2]	肖晨亮, 孙丽君, 何晓清, 徐路, 赵徐达. 三维打印+膜诱导技术修复重建下颌骨节段性缺损1例报道[J]. 《口腔颌面外科杂志》, 2023, 33(6): 418-423.
[3]	孙昊, 林超, 马旭彤, 仲若恺, 范欣. 矿化胶原联合浓缩生长因子治疗颌骨囊肿术后骨缺损的临床研究[J]. 《口腔颌面外科杂志》, 2023, 33(5): 320-325.
[4]	刘宪光, 颜淑云, 王旭霞. 牙骨质-骨结构不良21例诊治分析[J]. 《口腔颌面外科杂志》, 2023, 33(5): 330-335.
[5]	蔡永静, 陈志方, 张薇. 下颌舌侧孔解剖特点的CBCT观察[J]. 《口腔颌面外科杂志》, 2023, 33(4): 248-253.
[6]	吴秋裕, 陈浩, 朴正国, 张君伟, 欧阳可雄. 开窗减压术联合塞治器治疗混合牙列期颌骨囊肿1例[J]. 《口腔颌面外科杂志》, 2023, 33(4): 269-271.
[7]	康献刚, 牛军强, 赵冬霞, 霍霁. 重组人源肿瘤坏死因子受体融合蛋白对大鼠下颌骨缺损的干预效果[J]. 《口腔颌面外科杂志》, 2023, 33(2): 83-88.
[8]	宋丽娜, 吴春月, 秦清岩, 楚天舒, 刘麒麟. 不同类型血管化游离腓骨瓣修复下颌骨缺损的回顾性分析[J]. 《口腔颌面外科杂志》, 2023, 33(2): 104-110.
[9]	郭凌燕, 吴平凡, 陈芬, 李羽, 雷振革, 陈林林. 颌骨单纯性骨囊肿的临床分析[J]. 《口腔颌面外科杂志》, 2023, 33(1): 36-41.
[10]	张庆福. 3D打印钛合金植入体下颌骨修复重建远期效果观察1例[J]. 《口腔颌面外科杂志》, 2023, 33(1): 57-61.
[11]	张舒萌, 张琪, 马东洋. 髁突骨软骨瘤的临床诊疗现状与进展[J]. 《口腔颌面外科杂志》, 2022, 32(6): 380-382.
[12]	曾威, 姚麟, 郭萌萌, 邓璋. 虚拟手术及数字化手术导板在腓骨肌皮瓣重建下颌骨缺损中的应用:附17例报告[J]. 《口腔颌面外科杂志》, 2022, 32(6): 386-390.
[13]	孟志兵, 胡俊, 刘伟, 袁保锋, 邵苗苗, 高娟娟, 王恒俊. 三维打印技术在眶颧颌骨折术中的临床应用[J]. 《口腔颌面外科杂志》, 2022, 32(5): 303-308.
[14]	刘冬晓, 王伟. 下颌骨发育性缺损7例影像学分析[J]. 《口腔颌面外科杂志》, 2022, 32(5): 319-323.
[15]	孙国文, 田美. 药物相关性颌骨坏死不同阶段治疗方法述评[J]. 《口腔颌面外科杂志》, 2022, 32(3): 135-143.

模态框（Modal）标题

选择文件类型/文献管理软件名称

选择包含的内容

成年男性累及多处骨的朗格汉斯细胞组织细胞增生症1例

Langerhans cell histiocytosis involving multiple bones in an adult male: A case report

RichHTML

PDF

可视化

被引次数

摘要/Abstract

引用本文

使用本文

图/表 6

参考文献 15

相关文章 15

编辑推荐

Metrics

本文评价