儿童习惯性髋关节脱位病例报告及系统文献复习

Case report of habitual dislocation of the hip in children and a review of systematic literature

目的探讨观察加心理干预、石膏或支具固定以及手术治疗对儿童习惯性髋关节脱位(habitual dislocation of the hip,HDH)的临床疗效;采用系统文献复习的方法,探索HDH流行病学特征和维持髋关节稳定的治疗方案。方法回顾性分析2007年3月至2021年3月治疗并获随访的11例(12髋)HDH患儿相关资料,其中男2例,女9例,首次脱位年龄2.25(1.66,3.75)岁,确诊时年龄4.33(3.33,6.17)岁。同时检索1932年至2022年相关文献,并依据纳入和排除标准确认24篇HDH个案和临床报道纳入研究,获得文献中33例(38髋)HDH患儿资料,男5例,女28例,首次脱位年龄2.00(1.50,2.00)岁,确诊时年龄4.00(2.55,5.00)岁。分析共计44例(50髋)HDH患儿性别、首次脱位年龄、确诊时年龄、激发脱位方式、脱位侧别和方向、伴随症状、家族史、外伤史、影像学检查等数据获得HDH流行病学特征。对41例HDH患儿(3例缺少治疗描述,被剔除),采取观察加心理干预19例(46.34%),石膏或支具固定13例(31.71%),手术治疗9例(21.95%)。对治疗前后骨盆正位X线片和髋关节脱位瞬间"真空现象"采用AutoCAD软件测量颈干角、髋臼指数、中心边缘角(central edge angle,CE角)、Reimers不稳定指数。采用Harris标准评价髋关节功能,同时记录脱位消失时间。结果44例(50髋)HDH患儿随访时间(4.05±2.93)年,治疗后脱位消失的时间(1.28±1.19)年。其中女37例,男7例;首次脱位年龄(2.64±1.54)岁,确诊时年龄(4.52±2.64)岁;右侧脱位30例,左侧8例,双侧6例;屈髋、内收、内旋或屈髋、内收作为激发脱位方式39例;后位脱43例;脱位时伴有响亮的"咔哒"声41例;无痛性脱位36例;脱位瞬间捕捉到真空现象37例。所有患儿均无特定家族史和明显外伤史。观察加心理干预组、石膏或支具固定组和手术治疗组患儿一般资料差异无统计学意义(P>0.05)。患侧脱位瞬间Harris标准评价髋关节功能、CE角和Reimers不稳定指数与治疗前和治疗后比较,差异有统计学意义(H=127.51,P<0.001;H=55.70,P<0.001;H=54.69,P<0.001)。与手术治疗组术后脱位即刻消失相比较,观察加心理干预组和石膏或支具固定组脱位消失时间明显延长,差异有统计学意义(H=20.83,P<0.001)。结论无特定家族史和明显外伤的幼儿期女孩,无痛性髋关节后脱位伴"咔哒"声以及脱位瞬间真空现象是HDH显著的流行病学特征;观察加心理干预、石膏或支具固定建议作为初始治疗方案,当保守治疗无效,需手术治疗迅速稳定髋关节。

Objective To explore the clinical effect of observation and psychological intervention,splint or brace fixation as well as surgical treatment on habitual dislocation of the hip(HDH)and to combine the authors'data with a compilation of the cases from the literature,evaluingating the epidemiological characteristics of HDH and the treatment scheme to maintain the stability of hip joint by systematic literature review in children.Methods A retrospective analysis of the relevant data of 11 patients(12 hips)with HDH were treated from March 2007 to March 2021,including 2 boys and 9 girls.The age of the first dislocation was 2.25(1.66,3.75)years old and 4.33(3.33,6.17)years old at the age of diagnosis.At the same time,the relevant literature reports were searched from 1932 to 2022,and 24 HDH patients reports and clinical studies were confirmed to be included in this study according to the inclusion and exclusion criteria.The data of 33 patients(38 hips)with HDH who were obtained in the literature,including 5 boys and 28 girls.The age of the first dislocation was 2.00(1.50,2.00)years old and 4.00(2.55,5.00)years old at the age of diagnosis.A total of 44 patients(50 hips)with HDH who were analyzed,including gender,age of first dislocation,age of diagnosis,mode of stimulating dislocation,side and direction of dislocation,accompanying symptoms,family history,trauma history and imaging examination.The data of 41 patients with HDH(3 patients were excluded due to lack of treatment description)were treated with observation and psychological intervention in 19 patients,splint or brace fixation in 13 patients,and surgical treatment in 9 patients.The femoral neck-stem angle,acetabular index,central edge angle(CE angle)and Reimers instability index were measured by AutoCAD software,and the hip function was evaluated by Harris standard.Meantime,the disappearance time of dislocation was recorded.Results The average follow-up time of 44 patients(50 hips)with HDH were 4.05±2.93 years old,and the time of disappearance of dislocation after treatment were 1.28±1.19 years old.Patients were diagnosed as 7 boys and 37 girls,30 right and 8 left as well as 6 bilateral,the age of the first dislocation was 2.64±1.54 years old and 4.52±2.64 years old at the age of diagnosis.The data of 39 patients took hip flexion,adduction,internal rotation or hip flexion and adduction as the mode of stimulating dislocation.There were 43 patients with posterior dislocation,41 patients with an audible"click"sound during dislocation,36 patients with painless dislocation,and 37 patients with"vacuum phenomenon"were captured at the moment of dislocation.All patients with HDH had no specific family history and obvious history of trauma.There was no significant difference in general data between observation and psychological intervention group,splint or brace fixation group as well as surgical treatment group(P>0.05).Harris standard to evaluate hip function,CE angle and Reimers instability index of the affected side were significantly different from those before and after treatment at the moment of dislocation(H=127.51,P<0.001;H=55.70,P<0.001;H=54.69,P<0.001).Compared with the immediate disappearance of dislocation in the surgical treatment group,the disappearance time of dislocation in the observation and psychological intervention group and the splint or brace fixation group were significantly longer,and the difference was statistically significant(H=20.83,P<0.001).Conclusion Without specific family history and obvious trauma at young girls,painless posterior dislocation of hip with an audible"click"sound and"vacuum phenomenon"at the moment of dislocation are the significant epidemiological characteristics of HDH.Observation and psychological intervention,splint or brace fixation are recommended as the initial treatment scheme.When conservative treatment is ineffective,surgical treatment is needed to stabilize the hip joint quickly.