Duan Yaqin,Wu Shuihua,Hu Jihong,et al.Analysis of influencing factors for postoperative neurological functional outcomes in children with craniosynostosis and neurodevelopmental disorders[J].Journal of Clinical Pediatric Surgery,2025,(02):179-183.[doi:10.3760/cma.j.cn101785-202312027-013]
颅缝早闭合并神经发育障碍患儿手术后康复干预效果的影响因素分析
- Title:
- Analysis of influencing factors for postoperative neurological functional outcomes in children with craniosynostosis and neurodevelopmental disorders
- Keywords:
- Craniosynostosis; Rehabilitative Intervention; Neurodevelopmental Disorders; Influencing Factors
- 摘要:
- 目的 探讨颅缝早闭(craniosynostosis,CS)合并神经发育障碍患儿手术后康复干预效果的影响因素。方法 回顾性分析2016年9月至2023年3月于湖南省儿童医院接受手术治疗并行手术后康复干预的颅缝早闭合并神经发育障碍患儿临床资料。根据手术后首次及末次随访时格赛尔发育诊断量表(gesell developmental schedule,GDS)中发育商(developmental quotient,DQ)评分差值,评价CS合并神经发育障碍患儿手术后康复干预效果及其影响因素。将随访末次与首次GDS的DQ评分差值≥10分者归为效果良好组,DQ差值<10分者归为效果欠佳组。收集两组患儿性别、手术年龄、CS类型、主要带养者学历、术后首次及末次GDS评估年龄、康复干预起始年龄、康复干预总时长以及随访时长等,分析CS合并神经发育障碍患儿手术后康复干预效果的影响因素。结果 本研究共纳入41例颅缝早闭合并神经发育障碍患儿,其中效果欠佳组14例,效果良好组27例。单因素分析发现,两组术后首次DQ值[44.50(18.75,62.25)比69.00(62.00,78.00),Z=-3.259,P=0.001]、康复干预起始年龄[(28.00(17.5,36.75)个月比14.00(9.00,18.00)个月,Z=-3.358,P=0.000]、康复干预总时长[(7.500(2.50,12.75)个月比14.00(12.00,18.00)个月,Z=-2.604,P=0.008],差异均具有统计学意义(P<0.05)。多因素Logistic回归分析结果显示,术后首次DQ值(OR=1.140,95%CI:1.044~1.246)、康复干预起始年龄(OR=0.919,95%CI:0.849~0.996)、康复干预总时长(OR=1.314,95%CI:1.016~1.698)是CS合并神经发育障碍患儿手术后康复效果的独立影响因素。根据cut-off值将患儿分组为术后首次DQ≥52.5分组与＜52.5分组、康复干预总时长≥9.5个月组与＜9.5个月组、康复干预起始年龄≥22.5个月组与＜22.5个月组,各分组患儿DQ差值比较,差异均具有统计学意义(P<0.05)。结论 术后首次DQ评分≥52.5分较＜52.5分,康复干预总时长≥9.5个月较＜9.5个月,康复干预起始年龄≥22.5个月较＜22.5个月,康复效果好;提示CS合并神经发育障碍患儿手术后神经障碍程度越轻,康复干预时间越早,疗程越长,康复效果越好。
- Abstract:
- Objective To explore the influencing factors for the effectiveness of postoperative rehabilitative interventions in children with craniosynostosis (CS) plus neurodevelopmental disorders. Methods Retrospective analysis was conducted for the relevant clinical data of 41 CS children plus neurodevelopmental disorders undergoing postoperative rehabilitative interventions from September 2016 to March 2023.The influencing factors for the effectiveness of postoperative rehabilitative interventions were evaluated based upon the differential scores of developmental quotient (DQ) from the first and last assessments using Gesell Developmental Schedule (GDS).They were classified into a "good efficacy group" (n=27) if DQ score difference between the first and last GDS assessments was ≥10 points and a "poor efficacy group" (n=14) if DQ score difference <10 points.The relevant data collected for both groups included gender,age at surgery,type of CS,education level of primary caregivers,first and last GDS DQ scores,assessment age,age at the start of rehabilitative intervention,total duration of rehabilitation and follow-up time.The influencing factors for postoperative rehabilitation were then examined. Results Univariate analysis of two groups revealed statistically significant differences in postoperative first DQ score [44.50(18.75,62.25) vs.69.00(62.00,78.00),Z=-3.259,P=0.001],age at the start of rehabilitative intervention [28.00(17.5,36.75) vs.14.00(9.00,18.00) month,Z=-3.358,P=0.000]and total duration of rehabilitation [7.500(2.50,12.75) vs.14.00 (12.00,18.00) month,Z=-2.604,P=0.008](P<0.05).These variables were then included in multivariate Logistic regression analysis.The results indicated that postoperative first DQ score (OR=1.140,95%CI:1.044-1.246),age at the start of rehabilitative intervention (OR=0.919,95%CI:0.849-0.996) and total rehabilitative duration (OR=1.314,95%CI:1.016-1.698) were independent influencing factors for the effectiveness of postoperative rehabilitation.Based upon the cut-off values,comparisons of DQ score differences were made between two groups:postoperative first DQ ≥52.5 vs. <52.5,total rehabilitative duration ≥9.5 vs. <9.5 month and age at the start of rehabilitative intervention ≥22.5 vs. <22.5 month.The differences were statistically significant (P<0.05). Conclusions Group with postoperative first DQ ≥52.5 had better rehabilitative outcomes than group with DQ <52.5.Similarly,group with total rehabilitative duration ≥ 9.5 month had better outcomes than group with <9.5 month of rehabilitation and group with rehabilitation start age ≥ 22.5 month showed better outcomes than group aged <22.5 month.The milder postoperative neurodevelopmental impairment,the earlier rehabilitative intervention,the longer rehabilitative duration and the better rehabilitative outcomes.
参考文献/References:
[1] Thiele-Nygaard AE,Foss-Skiftesvik J,Juhler M.Intracranial pre-ssure,brain morphology and cognitive outcome in children with sagittal craniosynostosis[J].Childs Nerv Syst,2020,36(4):689-695.DOI:10.1007/s00381-020-04502-z.
[2] Junn AH,Long AS,Hauc SC,et al.Long-term neurocognitive outcomes in 204 single-suture craniosynostosis patients[J].Childs Nerv Syst,2023,39(7):1921-1928.DOI:10.1007/s00381-023-05908-1.
[3] 李婕,韩馨瑶,袁青骋,等.Gesell发育量表言语能力测试部分的语言学原理及本土化建议[J].中国听力语言康复科学杂志,2023,21(3):333-336.DOI:10.3969/j.issn.1672-4933.2023.03.028. Li J,Han XY,Yuan QC,et al.Linguistic rationale for verbal capability test section of Gesell developmental schedules and suggestions for localization[J].Chin Sci J Hear Speech Rehabil,2023,21(3):333-336.DOI:10.3969/j.issn.1672-4933.2023.03.028.
[4] Knudsen EI.Sensitive periods in the development of the brain and behavior[J].J Cogn Neurosci,2004,16(8):1412-1425.DOI:10.1162/0898929042304796.
[5] Gao W,Lin WL,Grewen K,et al.Functional connectivity of the infant human brain:plastic and modifiable[J].Neuroscientist,2017,23(2):169-184.DOI:10.1177/1073858416635986.
[6] 中国康复医学会儿童康复专业委员会,中国残疾人康复协会小儿脑性瘫痪康复专业委员会,中国医师协会康复医师分会儿童康复专业委员会,等.中国脑性瘫痪康复指南(2022)第二章:脑性瘫痪高危儿的评定与干预[J].中华实用儿科临床杂志,2022,37(13):974-982.DOI:10.3760/cma.j.cn101070-20220519-00574. Pediatric Rehabilitation Specialty Committee,Chinese Association of Rehabilitation Medicine; Rehabilitation Specialty Committee for Cerebral Palsy,Chinese Association of Rehabilitation of Disabled Persons; Pediatric Rehabilitation Specialty Committee,Chinese Medical Doctor Association,et al:Chinese Rehabilitation Guidelines for Cerebral Palsy (2022) Chapter II:Assessment and Intervention Guideline for Infants at An Elevated Risk of Cerebral Palsy[J].Chin J Appl Clin Pediatr,2022,37(13):974-982.DOI:10.3760/cma.j.cn101070-20220519-00574.
[7] Lidzba K,Küpper H,Kluger G,et al.The time window for successful right-hemispheric language reorganization in children[J].Eur J Paediatr Neurol,2017,21(5):715-721.DOI:10.1016/j.ejpn.2017.06.001.
[8] Wang D,Bian LZ,Hao XY,et al.Early rehabilitation after craniosynostosis surgery[J].Comput Mater Contin,2022,72(2):3899-3912.DOI:10.32604/cmc.2022.026660.
[9] Timberlake AT,McGee S,Allington G,et al.De novo variants implicate chromatin modification,transcriptional regulation,and retinoic acid signaling in syndromic craniosynostosis[J].Am J Hum Genet,2023,110(5):846-862.DOI:10.1016/j.ajhg.2023.03.017.
[10] Iwasa M,Shimizu Y,Sasayama D,et al.Twenty-year longitudinal birth cohort study of individuals diagnosed with autism spectrum disorder before seven years of age[J].J Child Psychol Psychiatry,2022,63(12):1563-1573.DOI:10.1111/jcpp.13614.
相似文献/References:
[1]伍添,吴水华.NFIA基因突变致脑畸形伴或不伴尿道缺陷疾病诊治分析[J].临床小儿外科杂志,2021,20(09):866.[doi:10.12260/lcxewkzz.2021.09.013]
Wu Tian,Wu Shuihua.Brain malformation with or without urethral defect due to NFIA gene mutation: one case report with a literature review[J].Journal of Clinical Pediatric Surgery,2021,20(02):866.[doi:10.12260/lcxewkzz.2021.09.013]
[2]吴水华,梁大中.非综合征性颅缝早闭患儿手术后ASQ-3年龄与发育进程问卷评估结果的相关因素分析[J].临床小儿外科杂志,2023,22(08):762.[doi:10.3760/cma.j.cn101785-202304003-012]
Wu Shuihua,Liang Dazhong.Analysis of factors related to ASQ-3 age and developmental process assessment questionnaire in children with nonsyndromic craniosynostosis after surgery[J].Journal of Clinical Pediatric Surgery,2023,22(02):762.[doi:10.3760/cma.j.cn101785-202304003-012]
[3]李制一,汪婧怡,顾硕.颅缝早闭的诊治现状及未来研究方向[J].临床小儿外科杂志,2024,(02):101.[doi:10.3760/cma.j.cn101785-202401007-001]
Li Zhiyi,Wang Jingyi,Gu Shuo.Current status of diagnosing and treating craniosynostosis and future research directions[J].Journal of Clinical Pediatric Surgery,2024,(02):101.[doi:10.3760/cma.j.cn101785-202401007-001]
[4]刘雨桐,曾高.不同年龄段矢状缝早闭的手术治疗探讨[J].临床小儿外科杂志,2024,(02):109.[doi:10.3760/cma.j.cn101785-202312012-002]
Liu Yutong,Zeng Gao.Surgical management of sagittal synostosis at different ages[J].Journal of Clinical Pediatric Surgery,2024,(02):109.[doi:10.3760/cma.j.cn101785-202312012-002]
[5]张迪,葛明,马文平,等.π型截骨术后辅助头盔治疗与颅骨重建术治疗婴幼儿非综合征型矢状缝早闭的疗效比较[J].临床小儿外科杂志,2024,(02):113.[doi:10.3760/cma.j.cn101785-202312054-003]
Zhang Di,Ge Ming,Ma Wenping,et al.A comparison of π craniectomy plus orthopedic helmet versus cranial vault remodeling for non-syndromic premature sagittal suture in infants[J].Journal of Clinical Pediatric Surgery,2024,(02):113.[doi:10.3760/cma.j.cn101785-202312054-003]
[6]李森,詹琪佳,肖波,等.额眶前移手术对额缝早闭患儿额叶容量及社会情绪改善的效果评价[J].临床小儿外科杂志,2024,(02):119.[doi:10.3760/cma.j.cn101785-202311041-004]
Li Sen,Zhan Qijia,Xiao Bo,et al.Improvements of frontal lobe volume and social emotions on metopic synostosis with frontal-orbital advancement[J].Journal of Clinical Pediatric Surgery,2024,(02):119.[doi:10.3760/cma.j.cn101785-202311041-004]
[7]许新科,林浩铭,林炜,等.儿童颅缝早闭二次手术影响因素分析[J].临床小儿外科杂志,2024,(02):125.[doi:10.3760/cma.j.cn101785-202312002-005]
Xu Xinke,Lin Haoming,Lin Wei,et al.Risk factors of secondary operation for craniosynostosis in children[J].Journal of Clinical Pediatric Surgery,2024,(02):125.[doi:10.3760/cma.j.cn101785-202312002-005]
[8]吴水华,Dae-jung Yang.以健侧为镜像治疗单侧冠状缝早闭症疗效分析[J].临床小儿外科杂志,2024,(02):130.[doi:10.3760/cma.j.cn101785-202312004-006]
Wu Shuihua,Dae-jung Yang.Efficacy of unilateral coronal craniostenosis using healthy side as a mirror image[J].Journal of Clinical Pediatric Surgery,2024,(02):130.[doi:10.3760/cma.j.cn101785-202312004-006]
[9]米伟阳,史航宇.颅缝早闭的分子机制与诊疗进展[J].临床小儿外科杂志,2025,(01):91.[doi:10.3760/cma.j.cn101785-202212024-017]
Mi Weiyang,Shi Hangyu.Molecular mechanisms and advances in diagnosis and treatment of craniosynostosis[J].Journal of Clinical Pediatric Surgery,2025,(02):91.[doi:10.3760/cma.j.cn101785-202212024-017]
备注/Memo
收稿日期:2023-12-12。
基金项目:湖南省残疾人康复科研项目(2021XK0221);湖南省科技厅临床医学研究中心项目(2021SK4018);湖南省自然科学基金资助项目(2025JJ50682)
通讯作者:吴水华,Email:292454021@qq.com