Ma Yue,Wang Shan,Li Changchun,et al.Single-center analysis of clinical features and prognosis of neuroblastoma children with bone metastasis[J].Journal of Clinical Pediatric Surgery,2022,21(02):115-120.[doi:10.3760/cma.j.cn.101785-202105011-004]
神经母细胞瘤并骨转移患儿临床特征及预后相关因素的单中心分析
- Title:
- Single-center analysis of clinical features and prognosis of neuroblastoma children with bone metastasis
- 关键词:
- 神经母细胞瘤/并发症; 神经母细胞瘤/分类; 肿瘤转移; 预后; 儿童
- Keywords:
- Neuroblastoma/CO; Neuroblastoma/CL; Neoplasm Metastasis; Prognosis; Child
- 摘要:
- 目的 分析伴骨转移神经母细胞瘤（neuroblastoma，NB）患儿的临床特征及预后相关因素，总结临床诊疗经验，以提高伴骨转移NB患儿的生存率。方法 以2013年12月至2020年12月重庆医科大学附属儿童医院肿瘤外科收治的伴骨转移NB患儿为研究对象，收集并总结患儿临床资料及预后情况。随访时间截至2021年3月31日。结果 共收集97例NB患儿，男68例，女29例，男女比例为2.4：1；中位年龄为49.4个月。首发症状：发热47例（48.5%），骨痛38例（39.2%），腹痛或腹胀29例（29.9%），咳嗽15例（15.5%）；伴骨转移的NB患儿存在多类骨转移（62.9%），且合并骨髓转移（73.2%），整体预后差，1年生存率为93.6%，5年生存率仅20.2%。经生存分析发现，女性、伴多类骨转移、肿瘤位于腹部、首诊时LDH测定值大于660 U/L以及术中肿瘤残留是预后不佳的影响因素，其中手术切除范围是独立预后影响因素。结论 伴骨转移的NB患儿临床表现多样，其预后受诸多因素影响，肿瘤复发或进展是主要的致死原因。对于此类患儿，建议术中尽可能完全切除肿瘤，以改善预后。
- Abstract:
- Objective To analyze the clinical features and prognostic factors of neuroblastoma (NB)children with bone metastasis.Methods The clinical data of 97 cases of NB children with bone metastasis treated in the Department of surgical Oncology, Children’s Hospital of Chongqing Medical University from December 2013 to December 2020 were retrospectively analyzed to compare the influence of different factors on the prognosis of the children.Results Among the 97 cases, there were 68 males and 29 females.The male to female ratio was 2.4:1, and the median age was 49.4 months.The common first symptoms were fever (48.5%), bone pain (39.2%), abdominal pain or abdominal distension (29.9%).The children with bone metastasis were prone to multiple bone metastasis (62.9%) and bone marrow metastasis (73.2%).The overall prognosis of NB children with bone metastasis was poor, with a 1-year overall survival rate of 93.6% and a 5-year overall survival rate of 20.2%.Survival analysis showed that female children, patients with multiple bone metastases, tumors located in the abdomen, LDH value greater than 660U/L at first diagnosis, and intraoperative tumor residue often suggested poor prognosis, among which surgical resection range was an independent prognostic factor.Conclusion NB children with bone metastasis have diverse clinical manifestations, and their prognosis is affected by many factors, and tumor recurrence or progression is the main cause of death.For such children, it is recommended that the tumor be removed as completely as possible intraoperatively to improve the prognosis.
参考文献/References:
[1] Utnes P,L?kke C,Fl?gstad T,et al.Clinically relevant biomarker discovery in high-risk recurrent neuroblastoma[J].Cancer Informatics,2019,18:1176935119832910.DOI:10.1177/1176935119832910.
[2] Pudela C,Balyasny S,Applebaum MA.Nervous system:embryonal tumors:neuroblastoma[J].Atlas Genet Cytogenet Oncol Haematol,2020,24(7):284-290.DOI:10.4267/2042/70771.
[3] Georgakis MK,Dessypris N,Baka M,et al.Neuroblastoma among children in Southern and Eastern European cancer registries:Variations in incidence and temporal trends compared to US[J].Int J Cancer,2018,142(10):1977-1985.DOI:10.1002/ijc.31222.
[4] Tas ML,Reedijk AMJ,Karim-Kos HE,et al.Neuroblastoma between 1990 and 2014 in the Netherlands:Increased incidence and improved survival of high-risk neuroblastoma[J].Eur J Cancer,2020,124:47-55.DOI:10.1016/j.ejca.2019.09.025.
[5] 刘登辉,唐湘莲,黄召,等.伴副肿瘤综合征儿童神经母细胞瘤临床特征分析[J].临床小儿外科杂志,2021,20(4):348-353.DOI:10.12260/lcxewkzz.2021.04.009. Liu DH,Tang XL,Huang Z,et al.Clinical features of neuroblastoma in children with concurrent paraneoplastic syndrome[J].J Clin Ped Sur,2021,20(4):348-353.DOI:10.12260/lcxewkzz.2021.04.009.
[6] 吕俊,席红卫,石正峰,等.儿童神经母细胞瘤误诊原因分析[J].肿瘤研究与临床,2020,32(3):199-201.DOI:10.3760/cma.j.cn115355-20190822-00375. Lu J,Xi HW,Shi ZF,et al.Analysis of misdiagnosis causes of neuroblastoma in children[J].Cancer Research And Clinic,2020,32(3):199-201.DOI:10.3760/cma.j.cn115355-20190822-00375.
[7] 隋晓东.儿童神经母细胞瘤29例诊断过程分析[J].中国现代医生,2020,58(34):18-22. Sui XD.Analysis of the diagnostic process of 29 cases of children neuroblastoma[J].China Modern Doctor,2020,58(34):18-22.
[8] Luksch R,Castellani MR,Collini P,et al.Neuroblastoma (Peripheral neuroblastic tumours)[J].Critical Reviews in Oncology/hematology,2016,107:163-181.DOI:10.1016/j.critrevonc.2016.10.001.
[9] Park JR,Eggert A,Caron H.Neuroblastoma:biology,prognosis,and treatment[J].Hematol Oncol Clin North Am,2010,24(1):65-86.DOI:10.1016/j.pcl.2007.10.014.
[10] 王国文,赵红宇.RANK/RANKL/OPG信号通路介导神经母细胞瘤骨侵袭/转移机制的研究进展[J].现代肿瘤医学,2017,25(5):813-816.DOI:10.3969/j.issn.1672-4992.2017.05.038. Wang GW,Zhao HY.Research progress of mechanism of bone invasion/metastasis in human neuroblastoma regulated by RANK/RANKL/OPG signaling pathway[J].J Mod Oncol,2017,25(5):813-816.DOI:10.3969/j.issn.1672-4992.2017.05.038.
[11] 张淑萍,沈乐园,宋一玲,等.ECT骨显像和骨髓涂片在神经母细胞瘤骨转移中的临床应用[J].现代肿瘤医学,2020,028(006):989-993.DOI:10.3969/j.issn.1672-4992.2020.06.025. Zhang SP,Shen LY,Song YL,et al.Clinical application of ECT bone imaging and bone marrow smear in bone metastasis of neuroblastoma[J].Modern oncology,2020,28(6):989-993.DOI:10.3969/j.issn.1672-4992.2020.06.025.
[12] 朱富艺,闫杰,曹嫣娜,等.血清神经元特异性烯醇化酶和尿香草扁桃酸与神经母细胞瘤临床病理特征的相关性[J].中国肿瘤临床,2019,46(22):28-34.DOI:10.3969/j.issn.1000-8179.2019.22.080. Zhun FY,Yan J,Cao YN,et al.Correlation of neuron-specific enolase and vanillylmandelic acid with clinico pathological features of neuroblastomas[J].Chin J Clin Oncol,2019,46(22):28-34.DOI:10.3969/j.issn.1000-8179.2019.22.080.
[13] Moroz V,Machin D,Hero B,et al.The prognostic strength of serum LDH and serum ferritin in children with neuroblastoma:A report from the International Neuroblastoma Risk Group (INRG) project[J].Pediatric Blood & Cancer,2020,67(8):e28359.DOI:10.1002/pbc.28359.
[14] Feriancikova B,Feglarova T,Krskova L,et al.NMYCMIAT Is an upstream regulator of NMYC and the disruption of the MIAT/NMYC axis induces cell death in amplified neuroblastoma cell lines[J].Int J Mol Sci,2021,22(7):3393.DOI:10.3390/ijms22073393.
[15] Otte J,Dyberg C,Pepich A,et al.MYCN Function in neuroblastoma development[J].Frontiers in Oncology,2021,10:624079.DOI:10.3389/fonc.2020.624079.
[16] 邢莉莉,吴晔明.MYCN与神经母细胞瘤相关性研究进展[J].临床小儿外科杂志,2014,13(4):338-340.DOI:10.3969/j.issn.1671-6353.2014.04.023. Xing LL,Wu YM.Research progress on the correlation between MYCN and neuroblastoma[J].J Clin Ped Sur,2014,13(4):338-340.DOI:10.3969/j.issn.1671-6353.2014.04.023.
[17] Fischer J,Pohl A,Volland R,et al.Complete surgical resection improves outcome in INRG high-risk patients with localized neuroblastoma older than 18months[J].Bmc Cancer,2017,17(1):520.DOI:10.1186/s12885-017-3493-0.
[18] Yeung F,Chung PH,Tam PK,et al.Is complete resection of high-risk stage Ⅳ neuroblastoma associated with better survival?[J].Journal of Pediatric Surgery,2015,50(12):2107-2111.DOI:10.1016/j.jpedsurg.2015.08.038.
[19] 郝腾,李斯慧,李兴军,等.伴骨转移神经母细胞瘤患儿的临床特征、治疗效果及预后[J].中华实用儿科临床杂志,2017,32(3):182-186.DOI:10.3760/cma.j.issn.2095-428X.2017.03.005. Hao T,Li SH,Li XJ,et al.Clinical features,treatment and prognosis of neuroblastoma with bone metastasis in children[J].Chin J Appl Clin Pediatr,2017,32(3):182-186.DOI:10.3760/cma.j.issn.2095-428X.2017.03.005.
[20] Pastor ER,Mousa SA.Current management of neuroblastoma and future direction[J].Crit Rev Oncol Hematol,2019,138:38-43.DOI:10.1016/j.critrevonc.2019.03.013.
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备注/Memo
收稿日期:2021-05-10。
基金项目:国家重点研发计划（2018YFC1313000）
通讯作者:王珊,Email:wangshan778@163.com