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治疗儿童长骨单纯性骨囊肿的干预措施。

Interventions for treating simple bone cysts in the long bones of children.

作者信息

Zhao Jia-Guo, Wang Jia, Huang Wan-Jie, Zhang Peng, Ding Ning, Shang Jian

机构信息

Department of Orthopaedic Surgery, Tianjin Hospital, No. 406 Jiefangnan Road, Hexi District, Tianjin, China, 300211.

Department of Paediatrics, Shengjing Hospital affiliated to China Medical University, No.36 Sanhao Street, Heping District, Shenyang, Liaoning, China, 110004.

出版信息

Cochrane Database Syst Rev. 2017 Feb 4;2(2):CD010847. doi: 10.1002/14651858.CD010847.pub3.

Abstract

BACKGROUND

Simple bone cysts, also known as a unicameral bone cysts or solitary bone cysts, are the most common type of benign bone lesion in growing children. Cysts may lead to repeated pathological fracture (fracture that occurs in an area of bone weakened by a disease process). Occasionally, these fractures may result in symptomatic malunion. The main goals of treatment are to decrease the risk of pathological fracture, enhance cyst healing and resolve pain. Despite the numerous treatment methods that have been used for simple bone cysts in long bones of children, there is no consensus on the best procedure. This is an update of a Cochrane review first published in 2014.

OBJECTIVES

To assess the effects (benefits and harms) of interventions for treating simple bone cysts in the long bones of children, including adolescents.We intended the following main comparisons: invasive (e.g. injections, curettage, surgical fixation) versus non-invasive interventions (e.g. observation, plaster cast, restricted activity); different categories of invasive interventions (i.e. injections, curettage, drilling holes and decompression, surgical fixation and continued decompression); different variations of each category of invasive intervention (e.g. different injection substances: autologous bone marrow versus steroid).

SEARCH METHODS

We searched the Cochrane Bone, Joint and Muscle Trauma Group Specialised Register, the Cochrane Central Register of Controlled Trials, MEDLINE, Embase, the China National Knowledge Infrastructure Platform, trial registers, conference proceedings and reference lists. Date of last search: April 2016.

SELECTION CRITERIA

Randomised and quasi-randomised controlled trials evaluating methods for treating simple bone cysts in the long bones of children.

DATA COLLECTION AND ANALYSIS

Two review authors independently screened search results and performed study selection. We resolved differences in opinion between review authors by discussion and by consulting a third review author. Two review authors independently assessed risk of bias and data extraction. We summarised data using risk ratios (RRs) or mean differences (MDs), as appropriate, and 95% confidence intervals (CIs). We used the Grading of Recommendations Assessment, Development, and Evaluation (GRADE) system to assess the overall quality of the evidence.

MAIN RESULTS

In this update in 2017, we did not identify any new randomised controlled trials (RCT) for inclusion. We identified one ongoing trial that we are likely to include in a future update. Accordingly, our results are unchanged. The only included trial is a multicentre RCT conducted at 24 locations in North America and India that compared bone marrow injection with steroid (methylprednisolone acetate) injection for treating simple bone cysts. Up to three injections were planned for participants in each group. The trial involved 90 children (mean age 9.5 years) and presented results for 77 children at two-year follow-up. Although the trial had secure allocation concealment, it was at high risk of performance bias and from major imbalances in baseline characteristics. Reflecting these study limitations, we downgraded the quality of evidence by two levels to 'low' for most outcomes, meaning that we are unsure about the estimates of effect. For outcomes where there was serious imprecision, we downgraded the quality of evidence by a further level to 'very low'.The trial provided very low quality evidence that fewer children in the bone marrow injection group had radiographically assessed healing of bone cysts at two years than in the steroid injection group (9/39 versus 16/38; RR 0.55 favouring steroid injection, 95% CI 0.28 to 1.09). However, the result was uncertain and may be compatible with no difference or small benefit favouring bone marrow injection. Based on an illustrative success rate of 421 children with healed bone cysts per 1000 children treated with steroid injections, this equates to 189 fewer (95% CI 303 fewer to 38 more) children with healed bone cysts per 1000 children treated with bone marrow injections. There was low quality evidence of a lack of difference between the two interventions at two years in functional outcome, based on the Activity Scale for Kids function score (0 to 100; higher scores equate to better outcome: MD -0.90; 95% CI -4.26 to 2.46) or in pain assessed using the Oucher pain score. There was very low quality evidence of a lack of differences between the two interventions for adverse events: subsequent pathological fracture (9/39 versus 11/38; RR 0.80, 95% CI 0.37 to 1.70) or superficial infection (two cases in the bone marrow group). Recurrence of bone cyst, unacceptable malunion, return to normal activities, and participant satisfaction were not reported.

AUTHORS' CONCLUSIONS: The available evidence is insufficient to determine the relative effects of bone marrow versus steroid injections, although the bone marrow injections are more invasive. Noteably, the rate of radiographically assessed healing of the bone cyst at two years was well under 50% for both interventions. Overall, there is a lack of evidence to determine the best method for treating simple bone cysts in the long bones of children. Further RCTs of sufficient size and quality are needed to guide clinical practice.

摘要

背景

单纯性骨囊肿,也称为单房性骨囊肿或孤立性骨囊肿,是儿童生长过程中最常见的良性骨病变类型。囊肿可能导致反复病理性骨折(即在因疾病过程而变弱的骨区域发生的骨折)。偶尔,这些骨折可能导致有症状的骨不连。治疗的主要目标是降低病理性骨折的风险,促进囊肿愈合并缓解疼痛。尽管已针对儿童长骨的单纯性骨囊肿使用了多种治疗方法,但对于最佳治疗方案尚无共识。这是对2014年首次发表的Cochrane系统评价的更新。

目的

评估治疗儿童(包括青少年)长骨单纯性骨囊肿的干预措施的效果(益处和危害)。我们计划进行以下主要比较:侵入性干预(如注射、刮除、手术固定)与非侵入性干预(如观察、石膏固定、限制活动);不同类别的侵入性干预(即注射、刮除、钻孔减压、手术固定和持续减压);每类侵入性干预的不同变体(如不同的注射物质:自体骨髓与类固醇)。

检索方法

我们检索了Cochrane骨、关节和肌肉创伤组专业注册库、Cochrane对照试验中央注册库、MEDLINE、Embase、中国知网、试验注册库、会议论文集和参考文献列表。最后一次检索日期:2016年4月。

入选标准

评估治疗儿童长骨单纯性骨囊肿方法的随机和半随机对照试验。

数据收集与分析

两位综述作者独立筛选检索结果并进行研究选择。我们通过讨论并咨询第三位综述作者来解决综述作者之间的意见分歧。两位综述作者独立评估偏倚风险并进行数据提取。我们酌情使用风险比(RRs)或均值差(MDs)以及95%置信区间(CIs)来汇总数据。我们使用推荐分级评估、制定和评价(GRADE)系统来评估证据的总体质量。

主要结果

在2017年的本次更新中,我们未识别出任何新的纳入随机对照试验(RCT)。我们识别出一项正在进行的试验,可能会纳入未来的更新中。因此,我们的结果未变。唯一纳入的试验是一项在北美和印度24个地点进行的多中心RCT,该试验比较了骨髓注射与类固醇(醋酸甲泼尼龙)注射治疗单纯性骨囊肿的效果。每组参与者计划最多进行三次注射。该试验涉及90名儿童(平均年龄9.5岁),并在两年随访时报告了77名儿童的结果。尽管该试验有可靠的分配隐藏,但存在较高的实施偏倚风险且基线特征存在重大不平衡。考虑到这些研究局限性,我们将大多数结局的证据质量下调两级至“低”,这意味着我们对效应估计不确定。对于存在严重不精确性的结局,我们将证据质量再下调一级至“极低”。该试验提供了极低质量的证据,表明骨髓注射组在两年时经影像学评估的骨囊肿愈合儿童少于类固醇注射组(9/39对16/38;RR 0.55,支持类固醇注射,95% CI 0.28至1.09)。然而,结果不确定,可能与无差异或支持骨髓注射的小益处相符。基于每1000名接受类固醇注射治疗的儿童中有421名骨囊肿愈合的说明性成功率,这相当于每1000名接受骨髓注射治疗的儿童中骨囊肿愈合的儿童少189名(95% CI少303名至多38名)。基于儿童活动量表功能评分(0至100;分数越高结局越好:MD -0.90;95% CI -4.26至2.46)或使用Oucher疼痛评分评估的疼痛,有低质量证据表明两种干预在两年时功能结局无差异。对于不良事件,有极低质量证据表明两种干预无差异:后续病理性骨折(9/39对11/38;RR 0.80,95% CI 0.37至1.70)或浅表感染(骨髓组两例)。未报告骨囊肿复发、不可接受的骨不连、恢复正常活动和参与者满意度。

作者结论

现有证据不足以确定骨髓注射与类固醇注射的相对效果,尽管骨髓注射侵入性更强。值得注意的是,两种干预在两年时经影像学评估的骨囊肿愈合率均远低于50%。总体而言,缺乏证据来确定治疗儿童长骨单纯性骨囊肿的最佳方法。需要进一步开展足够规模和质量的RCT来指导临床实践。

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