Feng Lei,Zhang Xuejun,Guo Dong,et al.Analysis of surgical efficacy for spinal vertebral damage in children with Langerhans cell histiocytosis[J].Journal of Clinical Pediatric Surgery,2021,20(08):761-765,772.[doi:10.12260/lcxewkzz.2021.08.011]
儿童脊柱朗格罕细胞组织细胞增生症患者手术疗效的回顾性分析
- Title:
- Analysis of surgical efficacy for spinal vertebral damage in children with Langerhans cell histiocytosis
- 关键词:
- 组织细胞肉瘤/外科学; 组织细胞肉瘤/药物疗法; 脊柱损伤/外科学; 脊髓损伤/康复; 治疗结果
- 分类号:
- R730.262.4;R687.37;R651.21
- 摘要:
- 目的 探讨手术治疗儿童朗格罕细胞组织细胞增生症（Langerhans cell histiocytosis，LCH）致脊柱椎体骨质破坏的疗效。方法 以2015年1月至2019年1月首都医科大学附属北京儿童医院收治的LCH导致脊柱椎体骨质破坏患者作为研究对象，均实施脊柱后路病灶刮除活检、椎弓根螺钉内固定术，术中全程予神经电生理监测（intraoperative neurophysiological monitoring，IONM），主要指标为体感诱发电位（somatosensory evoked potential，SEP）和运动诱发电位（motor evoked potential，MEP）监测波幅变化，于置钉前获得波幅基线。预警标准为：与基线相比，SEP波幅下降≥50%和（或）潜伏期延长≥10%，MEP波幅下降≥80%。术后病理检查确诊为LCH。均于术前及术后末次随访时拍摄全脊柱站立位正侧位X线片，并行脊柱CT检查，评估脊柱椎体病变部位节段性后凸角的大小以及病变椎体的高度。采用美国脊髓损伤协会（American Spinal Injury Association，ASIA）脊髓损伤水平评分量表评估患者术前、术后及末次随访时脊髓功能。结果 本研究共纳入67例LCH致脊柱椎体骨质破坏患者，男22例，女45例，平均年龄（4.4±2.6）岁，术后随访时间均在2年以上。手术前病变脊柱椎体存在节段性后凸角（30.1±10.1）°，手术后末次随访时下降至（5.9±5.1）°，差异有统计学意义（t=8.891，P<0.001）；手术前病变椎体高度（5.9±4.0） mm，末次随访时增加至（13.9±7.2） mm，差异有统计学意义（t=7.443，P<0.001）。67例中，53例术前无神经系统症状；14例存在下肢肌力减弱，ASIA分级为C或D级，围手术期神经功能损伤无明显恢复，至末次随访时ASIA分级均为E级。术中67例均能顺利引出SEP波幅，成功率100%；57例能引出MEP波幅，成功率85.1%，10例因下肢肌力下降明显，无法引出MEP波幅（14.9%）。结论 对于椎体破坏严重的LCH患者，尤其术前伴有神经症状者，脊柱后路病灶刮除活检、椎弓根螺钉内固定术可有效恢复椎体高度，促进患者神经功能的恢复。
- Abstract:
- Objective To explore the efficacy for spinal vertebral damage in children with Langerhans cell histiocytosis (LCH). Methods Retrospective analysis was conducted for 67 hospitalized LCH cases with vertebral destruction from January 2015 to January 2019. Posterior fixation and pathological biopsy were performed. During operation, somatosensory evoked potential (SEP) and motor evoked potential (MEP) were monitored simultaneously. Alterations of SEP amplitude decreasing >50% or latent period extending by 10% or MEP wave amplitude decreasing >80% were diagnosed as positive changes. Any type of myoelectric response hinting at provocation of nerve root was also interpreted as a positive change. Anteroposterior and lateral radiographs and computed tomography (CT) was employed for measuring sagittal kyphosis angle and vertebral height in vertebral lesions pre-operation and at the final follow-up. Perioperative neurological functions were evaluated by the scale of American Spinal Injury Association (ASIA). Results There were 22 boys and 45 girls with an average age of (4.4±2.6) years. The postoperative follow-up period was over 2 years. The segmental kyphosis angle declined post-operation (30.1±10.1 vs. 5.9±5.1). Significant differences existed in kyphosis angle between preoperative timepoints and the final follow-up (t=8.891, P<0.001). The mean vertebral height in lesion was significantly higher at the final follow-up than pre-operation (5.9±4.0 vs. 13.9±7.2 mm, t=7.443, P<0.001). And 53 of them were free of preoperative neurological symptoms with an ASIA grade of E while the remainders had reduced muscle strength of lower limbs with an ASIA grade of C/D. The results of neurological examination immediately post-operation were the same as those pre-operation. All children had an ASIA grade of E at the last follow-up. Success rate of SEP recording was 100% and MEP 85.1%. Elicitation of MEP failed (n=10, 14.9%). Conclusion For children with severe vertebral damage, especially those with preoperative neurological symptoms, surgery can not only elucidate the pathology, but also restore vertebral height, restore spinal stability and promote the recovery of damaged nerves
参考文献/References:
1 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.24367.
2 Huang WD, Yang XH, Wu ZP, et al. Langerhans cell histiocytosis of spine:a comparative study of clinical, imaging features, and diagnosis in children, adolescents, and adults[J]. Spine J, 2013, 13(9):1108-1117. DOI:10.1016/j.spinee.2013.03.013.
3 张颖, 李洪秋, 赵群, 等. 儿童骨嗜酸细胞性肉芽肿的诊断与治疗[J]. 临床小儿外科杂志, 2007, 6(1):23-25. DOI:10.3969/j.issn.1671-6353.2007.01.010. Zhang Y, Li HQ, Zhao Q, et al. The diagnostic and therapeutic method of eosinophilic granuloma of bone in children[J]. J Clin Ped Sur, 2007, 6(1):23-25. DOI:10.3969/j.issn.1671-6353.2007.01.010.
4 Denaro L, Longo UG, Papalia R, et al. Eosinophilic granuloma of the pediatric cervical spine[J]. Spine (Phila Pa 1976), 2008, 33(24):E936-E941. DOI:10.1097/BRS.0b013e3181859aab.
5 Emile JF, Abla O, Fraitag S, et al. Revised classification of histiocytoses and neoplasms of the macrophage-dendritic cell lineages[J]. Blood, 2016, 127(22):2672-2681. DOI:10.1182/blood-2016-01-690636.
6 Papadopoulou M, Panagopoulou P, Papadopoulou A, et al. The multiple faces of Langerhans cell histiocytosis in childhood:A gentle reminder[J]. Mol Clin Oncol, 2018, 8(3):489-492. DOI:10.3892/mco.2017.1539.
7 Maia RC, de Rezende LM, Robaina M, et al. Langerhans cell histiocytosis:differences and similarities in long-term outcome of paediatric and adult patients at a single institutional centre[J]. Hematology, 2015, 20(2):83-92. DOI:10.1179/1607845414Y.0000000173.
8 Sadashiva N, Rajalakshmi P, Mahadevan A, et al. Surgical treatment of Langerhans cell histiocytosis of cervical spine:case report and review of literature[J]. Childs Nerv Syst, 2016, 32(6):1149-1152. DOI:10.1007/s00381-015-2989-7.
9 Levine SE, Dormans JP, Meyer JS, et al. Langerhans’ cell histiocytosis of the spine in children[J]. Clin Orthop Relat Res, 1996, (323):288-293. DOI:10.1097/00003086-199602000-00040.
10 Rodallec MH, Feydy A, Larousserie F, et al. Diagnostic imaging of solitary tumors of the spine:what to do and say[J]. Radiographics, 2008, 28(4):1019-1041. DOI:10.1148/rg.284075156.
11 Han I, Suh ES, Lee SH, et al. Management of eosinophilic granuloma occurring in the appendicular skeleton in children[J]. Clin Orthop Surg, 2009, 1(2):63-67. DOI:10.4055/cios.2009.1.2.63.
12 王文莉, 肖兰福, 杨武森.小儿初期Langerhans细胞组织细胞增生症7例临床报告[J]. 临床小儿外科杂志, 2006, 5(2):146-148. DOI:10.3969/j.issn.1671-6353.2006.02.023. Wang WL, Xiao LF, Yang WS.Serial clinical report on early stage langerhans cell histiocytosi in children:a report of 7 cases[J]. J Clin Ped Sur, 2006, 5(2):146-148. DOI:10.3969/j.issn.1671-6353.2006.02.023.
13 Hung YC, Chang FC, Chen YW, et al. Langerhans’ cell histiocytosis in the pediatric spine:therapeutic dynamic change of spinal deformity[J]. Childs Nerv Syst, 2012, 28(8):1243-1250. DOI:10.1007/s00381-012-1764-2.
14 Bavbek M, Atalay B, Altinors N, et al. Spontaneous resolution of lumbar vertebral eosinophilic granuloma[J]. Acta Neurochir (Wien), 2004, 146(2):165-167. DOI:10.1007/s00701-003-0182-3.
15 Zheng Y, Ning B, Wu C, et al. Posterior focal curettage and spine stability reconstruction by temporary pedicle screw fixation in children with collapsed vertebrae due to eosinophilic granuloma[J]. Global Spine J, 2021:301282157. DOI:10.1177/2192568220988269.
16 Zhong N, Xu W, Meng T, et al. The surgical strategy for eosinophilic granuloma of the pediatric cervical spine complicated with neurologic deficit and/or spinal instability[J]. World J Surg Oncol, 2016, 14(1):301. DOI:10.1186/s12957-016-1063-6.
17 Tsai PY, Tzeng WS. Images in clinical medicine. Vertebra plana with spontaneous healing[J]. N Engl J Med, 2012, 366(19):e30. DOI:10.1056/NEJMicm1101582.
18 刘虎, 张学军, 曹隽, 等. 朗格罕细胞组织细胞增生症椎体破坏患儿手术治疗后脊柱及椎体形态改善情况研究[J]. 中华骨与关节外科杂志, 2017, 10(3):187-190. DOI:10.3969/j.issn.2095-9958.2017.03-02. Li F, Zhang XJ, Cao J, et al. Spinal and vertebral morphology improvement after surgical treatment of Langerhans cell histiocytosis with vertebral destruction in children[J]. Chinese Journal Bone and Joint Surgery, 2017, 10(3):187-190. DOI:10.3969/j.issn.2095-9958.2017.03-02.
备注/Memo
收稿日期:2021-07-06。
通讯作者:张学军,Email:zhang-x-j04@163.com