Jia-Guo Zhao1, Jia Wang1, Wan-Jie Huang2, Peng Zhang3, Ning Ding4, Jian Shang5. 1. Department of Orthopaedic Surgery, Tianjin Hospital, No. 406 Jiefangnan Road, Hexi District, Tianjin, China, 300211. 2. Department of Paediatrics, Shengjing Hospital affiliated to China Medical University, No.36 Sanhao Street, Heping District, Shenyang, Liaoning, China, 110004. 3. Department of Orthopaedic Surgery, The Second Affiliated Hospital of Soochow University, No 1055 Sanxiang Road, Suzhou, Jiangsu, China, 215004. 4. Department of Orthopaedic Surgery, Tanggu Traditional Chinese Medicine Hospital of Tianjin Binhai New Area, No. 90 Hangzhou Road, Tanggu District, Tianjin, China, 300451. 5. Department of Tuberculosis Clinics, Tianjin Center of Tuberculosis Control, No.124, Chifeng Road, Heping District, Tianjin, China, 300040.
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.
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.