山东大学耳鼻喉眼学报 ›› 2022, Vol. 36 ›› Issue (1): 55-59.doi: 10.6040/j.issn.1673-3770.0.2021.409

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婴儿中耳胚胎型横纹肌肉瘤1例并文献复习

李玲1,2,李聪1,2,孙岩2,陈良2   

  1. 1. 滨州医学院第二临床医学院, 山东 烟台 264003;
    2. 青岛大学附属烟台毓璜顶医院 耳鼻咽喉头颈外科/山东省耳鼻喉疾病临床医学研究中心, 山东 烟台 264000
  • 发布日期:2022-02-22
  • 通讯作者: 陈良. E-mail:entchenliang@hotmail.com
  • 基金资助:
    烟台市科技计划项目任务书(2020MSGY078)

A case of an infant with embryonal rhabdomyosarcoma in the middle ear and literature review

LI Ling1,2, LI Cong1,2, SUN Yan2, CHEN Liang2   

  1. 1. The second Medical College of BinZhou Medical University, Yantai 264003, Shandong, China;
    2. Department of Otorhinolaryngology & Head and Neck Surgery, Yantai Yuhuangding Hospital Affiliated to Qingdao University/ Shandong Provincial Clinical Research Center for Otorhinolaryngologic Diseases, Yantai 264000, Shandong, China
  • Published:2022-02-22

摘要: 目的 提高耳鼻喉科临床医师对婴儿中耳横纹肌肉瘤的认识。 方法 对1例婴儿中耳胚胎型横纹肌肉瘤进行临床资料分析并复习相关文献。 结果 患儿女,10月龄,患儿家长发现患儿左侧耳道肿物,伴耳道少量出血。CT及MRI提示左侧外耳道、鼓室及乳突内占位性病变,咽旁间隙、颈部、双侧腋窝淋巴结转移。通过活检手术后病理及免疫组化检查确诊为横纹肌肉瘤。患儿放化疗结束4个月后,因颅脑转移死亡。 结论 发生在婴儿中耳的横纹肌肉瘤少见,且位置隐匿,早期无典型临床表现,容易造成误诊及漏诊;该病恶性程度高,临床上应争取早发现、早诊断及早治疗,提高患者生存率。

关键词: 中耳, 胚胎型, 横纹肌肉瘤, 婴儿, 恶性肿瘤

Abstract: Objective Raising awareness of otolaryngologists on embryonal rhabdomyosarcoma disease. Methods The clinical data of an infant with embryonal rhabdomyosarcoma in the middle ear were analyzed, and related literatures were reviewed. Results The patient was a 10-month-old female with a bleeding mass in the left external auditory canal. Computed tomography and magnetic resonance imaging revealed soft tissue density shadows in the left external auditory meatus, tympanum, and mastoid sinus that were accompanied with an enlarged lymph node in the parapharyngeal, neck and bilateral axilla. Moreover, the diagnosis of RMS was confirmed by postoperative pathological and immunohistochemical examination after biopsy. The patient died of brain metastasis four months after the end of radiation and chemotherapy. Conclusion RMS in the middle ear is rare. Due to its obscure location and lack of typical clinical manifestations in the early stage, RMS is difficult to detect and prone to misdiagnosis. The malignant degree of this disease is high. Thus, the early detection, diagnosis, and treatment of this disease should be further studied to improve the survival rate of patients.

Key words: Middle ear, Embryonic, Rhabdomyosarcoma, Infant, Malignancy

中图分类号: 

  • R764.2
[1] 中国抗癌协会小儿肿瘤专业委员会, 中华医学会儿科学分会血液学组, 中华医学会小儿外科学分会肿瘤组. 中国儿童及青少年横纹肌肉瘤诊疗建议(CCCG-RMS-2016)[J]. 中华儿科杂志, 2017, 55(10): 724-728. doi:10.3760/cma.j.issn.0578-1310.2017.10.002.
[2] Hayes SM, Biggs TC, Bird JH, et al. Rhabdomyosarcoma of the middle ear cavity presenting with Horner's syndrome[J]. Annals, 2019, 101(1): e8-e10. doi:10.1308/rcsann.2018.0157.
[3] 何健, 钱立庭, 高劲. 外耳道腺样囊性癌侵犯中耳、颞叶且伴颈部淋巴结转移一例报告[J]. 山东大学耳鼻喉眼学报, 2018, 32(6): 109-111,118. doi: 10.6040/j.issn.1673-3770.0.2017.424. HE Jian, QIAN Liting, GAO Jin. A case of external auditory canal adenoid cystic carcinoma invading the middle ear and temporal lobe, with cervical lymph node metastasis[J]. J Otolaryngol Ophthalmol Shandong Univ, 2018,32(6): 109-111,118. doi: 10.6040/j.issn.1673-3770.0.2017.424.
[4] 赵笑冰, 张大为, 陈仁杰. 中耳间变性大 T 细胞淋巴瘤一例并文献复习[J/OL].山东大学耳鼻喉眼学报,2022[2022-02-18]. doi:10.6040/j.issn.1673-3770.0.2021.318. ZHAO Xiaobing, ZHANG Dawei, CHEN Renjie. A case of middle-ear anaplastic large T- cell lymphoma and literature review[J]. J Otolaryngol Ophthalmol Shandong Univ, 2022[2022-02-18]. doi:10.6040/j.issn.1673-3770.0.2021.318.
[5] Jo VY, Fletcher CDM. WHO classification of soft tissue tumours: an update based on the 2013(4th)edition[J]. Pathology, 2014, 46(2): 95-104. doi:10.1097/PAT.0000000000000050.
[6] Leiner J, Le Loarer F. The current landscape of rhabdomyosarcomas: an update[J]. Virchows Arch, 2020, 476(1): 97-108. doi:10.1007/s00428-019-02676-9.
[7] Fischer TD, Gaitonde SG, Bandera BC, et al. Pediatric-protocol of multimodal therapy is associated with improved survival in AYAs and adults with rhabdomyosarcoma[J]. Surgery, 2018, 163(2): 324-329. doi:10.1016/j.surg.2017.10.027.
[8] Janz TA, Camilon PR, Cheung AY, et al. A review of pediatric middle ear tumors and analysis of the demographics, management, and survival of pediatric rhabdomyosarcomas of the middle ear[J]. Int J Pediatr Otorhinolaryngol, 2018, 112: 109-112. doi:10.1016/j.ijporl.2018.06.017.
[9] 李克鹏, 刘柱, 金书丞, 等. 鼻部横纹肌肉瘤诊疗分析[J]. 临床耳鼻咽喉头颈外科杂志, 2017,31(18): 1398-1401,1407. doi:10.13201/j.issn.1001-1781.2017.18.005. LI Kepeng, LIU Zhu, JIN Shucheng, et al. Clinical analysis of nose rhabdomyosarcoma[J]. J Clin Otorhinolaryngol Head Neck Surg, 2017,31(18): 1398-1401,1407. doi:10.13201/j.issn.1001-1781.2017.18.005.
[10] Hicks J, Flaitz C. Rhabdomyosarcoma of the head and neck in children[J]. Oral Oncol, 2002, 38(5): 450-459. doi:10.1016/s1368-8375(01)00105-1.
[11] 彭晓敏, 黎阳, 熊稀霖, 等. 放疗及二次根治手术对儿童横纹肌肉瘤的疗效影响分析[J]. 中国小儿血液与肿瘤杂志, 2021,26(1): 29-34. doi: 10.3969/j.issn.1673-5323.2021.01.005. PENG Xiaomin, LI Yang, XIONG Xilin, et al. Effect of radiotherapy and second radicalsurgery on rhabdomyosarcoma in children[J]. J China Pediatr Blood Cancer, 2021,26(1): 29-34. doi: 10.3969/j.issn.1673-5323.2021.01.005.
[12] Dasgupta R, Fuchs J, Rodeberg D. Rhabdomyosarcoma[J]. Semin Pediatr Surg, 2016, 25(5): 276-283. doi:10.1053/j.sempedsurg.2016.09.011.
[13] Wharam MD, Hanfelt JJ, Tefft MC, et al. Radiation therapy for rhabdomyosarcoma: local failure risk for Clinical Group III patients on Intergroup Rhabdomyosarcoma Study II[J]. Int J Radiat Oncol Biol Phys, 1997, 38(4): 797-804. doi:10.1016/s0360-3016(97)00120-x.
[14] Lockney NA, Friedman DN, Wexler LH, et al. Late toxicities of intensity-modulated radiation therapy for head and neck rhabdomyosarcoma[J]. Pediatr Blood Cancer, 2016, 63(9): 1608-1614. doi:10.1002/pbc.26061.
[15] Hu JW, Liu SL, Qiu JH. Embryonal rhabdomyosarcoma of the middle ear[J]. Otolaryngol Head Neck Surg, 2002, 126(6): 690-692. doi:10.1067/mhn.2002.123922.
[16] Linabery AM, Ross JA. Childhood and adolescent cancer survival in the US by race and ethnicity for the diagnostic period 1975-1999[J]. Cancer, 2008, 113(9): 2575-2596. doi:10.1002/cncr.23866.
[17] Skapek SX, Ferrari A, Gupta AA, et al. Rhabdomyosarcoma[J]. Nat Rev Dis Primers, 2019, 5(1): 1-19. doi:10.1038/s41572-018-0051-2.
[18] Hibbitts E, Chi YY, Hawkins DS, et al. Refinement of risk stratification for childhood rhabdomyosarcoma using FOXO1 fusion status in addition to established clinical outcome predictors: a report from the Children's Oncology Group[J]. Cancer Med, 2019, 8(14): 6437-6448. doi:10.1002/cam4.2504.
[19] 中华医学会病理学分会儿科病理学组, 中国抗癌协会小儿肿瘤专业委员会病理学组, 福棠儿童医学发展研究中心病理专业委员会. 儿童横纹肌肉瘤病理诊断规范化专家共识[J]. 中华病理学杂志, 2021, 50(10): 1110-1115. doi:10.3760/cma.j.cn112151-20210112-00033. Consensus on the diagnosis of pediatric rhabdomyosarcoma[J]. Chinese Journal of Pathology, 2021, 50(10): 1110-1115. doi:10.3760/cma.j.cn112151-20210112-00033.
[20] Malempati S, Hawkins DS. Rhabdomyosarcoma: review of the Children's Oncology Group(COG)Soft-Tissue Sarcoma Committee experience and rationale for current COG studies[J]. Pediatr Blood Cancer, 2012, 59(1): 5-10. doi:10.1002/pbc.24118.
[21] 徐红艳, 黄慧, 杨文萍. 小儿耳鼻部胚胎性横纹肌肉瘤2例并文献复习[J]. 临床与实验病理学杂志, 2013, 29(4): 445-446. doi:10.13315/j.cnki.cjcep.2013.04.017.
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