Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Meta-Analysis
. 2019 Mar 7;3(3):CD010778.
doi: 10.1002/14651858.CD010778.pub2.

Intralesional treatment versus wide resection for central low-grade chondrosarcoma of the long bones

Edwin F Dierselhuis  1 , Krista A Goulding, Martin Stevens, Paul C Jutte
Affiliations
Meta-Analysis

Intralesional treatment versus wide resection for central low-grade chondrosarcoma of the long bones

Edwin F Dierselhuis et al. Cochrane Database Syst Rev. .

Abstract

Background: Grade I or low-grade chondrosarcoma (LGCS) is a primary bone tumour with low malignant potential. Historically, it was treated by wide resection, since accurate pre-operative exclusion of more aggressive cancers can be challenging and under-treatment of a more aggressive cancer could negatively influence oncological outcomes. Intralesional surgery for LGCS has been advocated more often in the literature over the past few years. The potential advantages of less aggressive treatment are better functional outcome and lower complication rates although these need to be weighed against the potential for compromising survival outcomes.

Objectives: To assess the benefits and harms of intralesional treatment by curettage compared to wide resection for central low-grade chondrosarcoma (LGCS) of the long bones.

Search methods: We searched the Cochrane Central Register of Controlled Trials (CENTRAL; 2018, Issue 4), MEDLINE and Embase up to April 2018. We extended the search to include trials registries, reference lists of relevant articles and review articles. We also searched 'related articles' of included studies suggested by PubMed.

Selection criteria: In the absence of prospective randomised controlled trials (RCTs), we included retrospective comparative studies and case series that evaluated outcome of treatment of central LGCS of the long bones. The primary outcome was recurrence-free survival after a minimal follow-up of 24 months. Secondary outcomes were upgrading of tumour; functional outcome, as assessed by the Musculoskeletal Tumor Society (MSTS) score; and occurrence of complications.

Data collection and analysis: We used standard methodological procedures recognised by Cochrane. We conducted a systematic literature search using several databases and contacted corresponding authors, appraised the evidence using the ROBINS-I risk of bias tool and GRADE, and performed a meta-analysis. If data extraction was not possible, we included studies in a narrative summary.

Main results: We included 18 studies, although we were only able to extract participant data from 14 studies that included a total of 511 participants; 419 participants were managed by intralesional treatment and 92 underwent a wide resection. We were not able to extract participant data from four studies, including 270 participants, and so we included them as a narrative summary only. The evidence was at high risk of performance, detection and reporting bias.Meta-analysis of data from 238 participants across seven studies demonstrated little or no difference in recurrence-free survival after intralesional treatment versus wide resection for central LGCS in the long bones (risk ratio (RR) 0.98; 95% confidence interval (CI) 0.92 to 1.04; very low-certainty evidence). MSTS scores were probably better after intralesional surgery (mean score 93%) versus resection (mean score 78%) with a mean difference of 12.69 (95% CI 2.82 to 22.55; P value < 0.001; 3 studies; 72 participants; low-certainty evidence). Major complications across six studies (203 participants) were lower in cases treated by intralesional treatment (5/125 cases) compared to those treated by wide resection (18/78 cases), with RR 0.23 (95% CI 0.10 to 0.55; low-certainty evidence). In four people (0.5% of total participants) a high-grade (grade 2 or dedifferentiated) tumour was found after a local recurrence. Two participants were treated with second surgery with no evidence of disease at their final follow-up and two participants (0.26% of total participants) died due to disease. Kaplan-Meier analysis of data from 115 individual participants across four studies demonstrated 96% recurrence-free survival after a maximum follow-up of 300 months after resection versus 94% recurrence-free survival after a maximum follow-up of 251 months after intralesional treatment (P value = 0.58; very low-certainty evidence). Local recurrence or metastases were not reported after 41 months in either treatment group.

Authors' conclusions: Only evidence of low- and very low-certainty was available for this review according to the GRADE system. Included studies were all retrospective in nature and at high risk of selection and attrition bias. Therefore, we could not determine whether wide resection is superior to intralesional treatment in terms of event-free survival and recurrence rates. However, functional outcome and complication rates are probably better after intralesional surgery compared to wide resection, although this is low-certainty evidence, considering the large effect size. Nevertheless, recurrence-free survival was excellent in both groups and a prospective RCT comparing intralesional treatment versus wide resection may be challenging for both practical and ethical reasons. Future research could instead focus on less invasive treatment strategies for these tumours by identifying predictors that help to stratify participants for surgical intervention or close observation.

PubMed Disclaimer

Conflict of interest statement

  1. ED: none known.

  2. KG: none known.

  3. MS: none known..

  4. PJ: none known.

ED, MS and PJ are authors of the included study Dierselhuis 2016.

Figures

1
1
Study flow diagram
2
2
'Risk of bias' summary: review authors' judgements about each risk of bias item for each included study
3
3
'Risk of bias' graph: review authors' judgements about each risk of bias item presented as percentages across all included studies
4
4
Forest plot of comparison 1. Comparative studies, outcome 1.1 recurrence‐free survival. Event = recurrence‐free survival
5
5
Forest plot of comparison 1. Comparative studies, outcome 1.2 function by MSTS score
6
6
Forest plot of comparison 1. Comparative studies, outcome 1.3 complications. Event = major complication (e.g. fracture, infection)
7
7
Kaplan Meyer survival curve of recurrence‐free survival of participants with LGCS in the long bones. P = 0.58
See this image and copyright information in PMC

Update of

  • doi: 10.1002/14651858.CD010778

References

References to studies included in this review

Aarons 2009 {published and unpublished data}
    1. Aarons C, Potter BK, Adams SC, Pitcher DJ, Templet TH. Extended intralesional treatment versus resection of low‐grade chondrosarcomas. Clinical Orthopaedics and Related Research 2009;467:2105–11. - PMC - PubMed
Bauer 1995 {published data only}
    1. Bauer HC, Brosjo O, Kreicbergs A, Lindholm J. Low risk of recurrence of enchondroma and low‐grade chondrosarcoma in extremities. 80 patients followed for 2 ‐ 25 years. Acta Orthopaedica Scandinavica 1995;66(3):283‐8. - PubMed
Campanacci 2013 {published data only (unpublished sought but not used)}
    1. Campanacci DA, Scoccianti G, Franchi A. Surgical treatment of central grade 1 chondrosarcoma of the appendicular skeleton. Journal of Orthopaedics and Traumatology 2013;14:101‐7. - PMC - PubMed
Chen 2017 {published data only (unpublished sought but not used)}
    1. Chen YC, Wu PK, Chen CF, Chen WM. Intralesional curettage of central low‐grade chondrosarcoma: a midterm follow‐up study. Journal of the Chinese Medical Association : JCMA 2017;80(3):178‐82. - PubMed
Dierselhuis 2016 {published data only (unpublished sought but not used)}
    1. Dierselhuis EF, Gerbers JG, Ploegmakers JJW, Stevens M, Suurmeijer AJH, Jutte PC. Local treatment with adjuvant therapy for central atypical cartilaginous tumors in the long bones. Journal of Bone and Joint Surgery 2016;98:303‐13. - PubMed
Di Giorgio 2011 {published data only (unpublished sought but not used)}
    1. Giorgio L, Touloupakis G, Vitullo F, Sodano L, Mastantuono M, Villani C. Intralesional curettage, with phenol and cement as adjuvants, for low‐grade intramedullary chondrosarcoma of the long bones. Acta Orthopaedica Belgica 2011;77:666‐9. - PubMed
Donati 2010 {published data only (unpublished sought but not used)}
    1. Donati D, Colangeli S, Colangeli M, Bella C, Bertoni F. Surgical treatment of grade I central chondrosarcoma. Clinical Orthopaedics and Related Research 2010;468(2):581‐9. - PMC - PubMed
Etchebehere 2005 {published data only (unpublished sought but not used)}
    1. Etchebehere M, Camargo OP, Croci AT, Oliveira CR, Baptista AM. Relationship between surgical procedure and outcome for patients with grade I chondrosarcomas. Clinics (Sao Paulo, Brazil) 2005;60:121‐6. - PubMed
Funovics 2010 {published data only (unpublished sought but not used)}
    1. Funovics PT, Panotopoulos J, Sabeti‐Aschraf M, Abdolvahab F, Funovics JM, Lang S, et al. Low‐grade chondrosarcoma of bone: experiences from the Vienna Bone and Soft Tissue Tumour Registry. International Orthopaedics 2011;35(7):1049‐56. - PMC - PubMed
Gunay 2013 {published data only (unpublished sought but not used)}
    1. Gunay C, Atalar H, Hapa O, Basarir K, Yildiz, Saglik Y. Surgical management of Grade I chondrosarcoma of the long bones. Acta Orthopaedica Belgica 2013;79:331‐7. - PubMed
Hanna 2009 {published data only (unpublished sought but not used)}
    1. Hanna SA, Whittingham‐Jones P, Sewell MD, Pollock RC, Skinner JA, Saifuddin A, et al. Outcome of intralesional curettage for low‐grade chondrosarcoma of long bones. Journal of Cancer Surgery 2009;35:1343‐47. - PubMed
Kim 2015 {published data only (unpublished sought but not used)}
    1. Kim W, Han I, Kim EJ, Kang S, Kim H. Outcomes of curettage and anhydrous alcohol adjuvant for low‐grade chondrosarcoma of long bone. Surgical Oncology 2015;24:89‐94. - PubMed
Kim 2018 {published data only (unpublished sought but not used)}
    1. Kim W, Lee JS, Chung HW. Outcomes after extensive manual curettage and limited burring for atypical cartilaginous tumour of long bone. Journal of Bone and Joint Surgery. British Volume 2018;100‐B(2):256‐61. - PubMed
Leerapun 2007 {published data only (unpublished sought but not used)}
    1. Leerapun T, Hugate RR, Inwards CY, Scully SP, Sim FH. Surgical management of conventional grade I chondrosarcoma of long bones. Clinical Orthopaedics and Related Research 2007;463:166‐72. - PubMed
Mermerkaya 2014 {published data only (unpublished sought but not used)}
    1. Mermerkaya MU, Bekmez S, Karaaslan F, Danisman M, Kosemehmetoglu K, Gedikoglu G, et al. Intralesional curettage and cementation for low‐grade chondrosarcoma of long bones: retrospective study and literature review. World Journal of Surgical Oncology 2014;12:336‐41. - PMC - PubMed
Mohler 2010 {published data only (unpublished sought but not used)}
    1. Mohler DG, Chiu R, McCall DA, Avedian RS. Curettage and cryosurgery for low‐grade cartilage tumors is associated with low recurrence and high function. Clinical Orthopaedics and Related Research 2010;10(468):2765‐73. - PMC - PubMed
Van der Geest 2008 {published data only (unpublished sought but not used)}
    1. Geest IC, Valk MH, Rooy JW, Pruszczynski M, Veth RP, Schreuder HW. Oncological and functional results of cryosurgical therapy of enchondromas and chondrosarcomas grade 1. Journal of Surgical Oncology 2008;98(6):421‐6. - PubMed
Verdegaal 2012 {published data only (unpublished sought but not used)}
    1. Verdegaal SH, Brouwers HF, Zwet EW, Hogendoorn PC, Taminiau AH. Low‐grade chondrosarcoma of long bones treated with intralesional curettage followed by application of phenol, ethanol, and bone‐grafting. Journal of Bone and Joint Surgery. American Volume 2012;94(13):1201‐7. - PubMed

References to studies excluded from this review

Ahlmann 2007 {published data only (unpublished sought but not used)}
    1. Ahlmann ER, Menendez LR, Fedenko AN, Learch T. Influence of cryosurgery on treatment outcome of low‐grade chondrosarcoma. Clinical Orthopaedics and Related Research 2006;451:201‐7. - PubMed
Errani 2017 {published data only (unpublished sought but not used)}
    1. Errani C, Tsukamoto S, Ciani G, Akahane M, Cevolani L, Tanzi P, et al. Risk factors for local recurrence from atypical cartilaginous tumour and enchondroma of the long bones. European Journal of Orthopaedic Surgery & Traumatology : Orthopedie Traumatologie 2017;27(6):805‐11. - PubMed
Lee 1999 {published data only (unpublished sought but not used)}
    1. Lee FY, Mankin HJ, Fondren G, Gebhardt MC, Springfield DS, Rosenberg AE, et al. Chondrosarcoma of bone: an assessment of outcome. Journal of Bone and Joint Surgery. American Volume 1999;81(3):326‐38. - PubMed
Okada 2009 {published data only}
    1. Okada K, Nagasawa H, Chida S, Nishida J. Curettage with pasteurization in situ for grade 1 chondrosarcoma ‐ long‐term follow up study of less invasive surgical procedure. Medical Science Monitor 2009;15(3):CS44‐8. - PubMed
Ozaki 1996 {published data only}
    1. Ozaki T, Lindner N, Hillmann A, Rödl R, Blasius S, Winkelmann W. Influence of intralesional surgery on treatment outcome of chondrosarcoma. Cancer 1996;77(7):1292‐7. - PubMed
Puri 2009 {published data only}
    1. Puri A, Shah M, Agarwal MG, Jambhekar NA, Basappa P. Chondrosarcoma of bone: does the size of the tumor, the presence of a pathologic fracture, or prior intervention have an impact on local control and survival?. Journal of Cancer Research and Therapeutics 2009;5(1):14‐9. - PubMed
Schreuder 1998 {published data only}
    1. Schreuder HW, Pruszczynski M, Veth RP, Lemmens JA. Treatment of benign and low‐grade malignant intramedullary chondroid tumours with curettage and cryosurgery. European Journal of Surgical Oncology 1998;24(2):120‐6. - PubMed
Souna 2010 {published data only}
    1. Souna BS, Belot N, Duval H, Langlais F, Thomazeau H. No recurrences in selected patients after curettage with cryotherapy for grade I chondrosarcomas. Clinical Orthopaedics and Related Research 2010;468(7):1956‐62. - PMC - PubMed

References to studies awaiting assessment

Andreou 2011 {published data only (unpublished sought but not used)}
    1. Andreou D, Ruppin S, Fehlberg S, Pink D, Werner M, Tunn PU. Survival and prognostic factors in chondrosarcoma results in 115 patients with long‐term follow‐up. Acta Orthopaedica 2011;82(6):749‐55. - PMC - PubMed
Angelini 2012 {published data only (unpublished sought but not used)}
    1. Angelini A, Guerra G, Mavrogenis AF, Pala E, Picci P, Ruggieri P. Clinical outcome of central conventional chondrosarcoma. Journal of Surgical Oncology 2012;106(8):929‐37. - PubMed
de Camargo 2010 {published data only (unpublished sought but not used)}
    1. Camargo OP, Baptista AM, Atanásio MJ, Waisberg DR. Chondrosarcoma of bone: lessons from 46 operated cases in a single institution. Clinical Orthopaedics and Related Research 2010;468(11):2969‐75. - PMC - PubMed
Ma 2009 {published data only}
    1. Ma XJ, Dong Y, Zhang CL, Zeng BF. Recurrence analysis in 66 cases with grade I and grade II chondrosarcomas in the extremities. Orthopaedic Surgery 2009;1(2):132‐6. - PMC - PubMed
Meftah 2013 {published data only (unpublished sought but not used)}
    1. Meftah M, Schult P, Henshaw RM. Long‐term results of intralesional curettage and cryosurgery for treatment of low‐grade chondrosarcoma. Journal of Bone and Joint Surgery. American Volume 2013;95(15):1358‐64. - PubMed
Streitbuerger 2009 {published data only (unpublished sought but not used)}
    1. Streitbürger A, Ahrens H, Balke M, Buerger H, Winkelmann W, Gosheger G, et al. Grade I chondrosarcoma of bone: the Münster experience. Journal of Cancer Research and Clinical Oncology 2009;135(4):543‐50. - PMC - PubMed

Additional references

Bjornsson 1998
    1. Bjornsson J, McLeod RA, Unni KK, Ilstrup DM, Pritchard DJ. Primary chondrosarcoma of long bones and limb girdles. Cancer 1998;83(10):2105‐19. - PubMed
Deeks 2001
    1. Deeks JJ, Altman DG, Bradburn MJ. Statistical methods for examining heterogeneity and combining results from several studies in meta‐analysis. Systematic Reviews in Health Care: Meta‐Analysis in Context. 2nd Edition. London: BMJ Publication Group, 2001.
Eefting 2009
    1. Eefting D, Schrage YM, Geirnaerdt MJ, Le Cessie S, Taminiau AH, Bovée JV, et al. Assessment of interobserver variability and histologic parameters to improve reliability in classification and grading of central cartilaginous tumors. American Journal of Surgical Pathology 2009;33(1):50‐7. - PubMed
Enneking 1986
    1. Enneking WF. A system of staging musculoskeletal neoplasms. Clinical Orthopaedics and Related Research 1986;204:9‐24. - PubMed
Enneking 1993
    1. Enneking WF, Dunham W, Gebhardt MC, Malawar M, Pritchard DJ. A system for the functional evaluation of reconstructive procedures after surgical treatment of tumors of the musculoskeletal system. Clinical Orthopaedics and Related Research 1993;286:241‐6. - PubMed
Eriksson 1980
    1. Eriksson AI, Schiller A, Mankin HJ. The management of chondrosarcoma of bone. Clinical Orthopaedics and Related Research 1980;153:44‐66. - PubMed
ESMO 2012
    1. ESMO/European Sarcoma Network Working Group. Bone sarcomas: ESMO Clinical Practice Guidelines for diagnosis, treatment and follow‐up. Annals of Oncology 2012;23(Suppl 7):vii100‐9. - PubMed
Evans 1977
    1. Evans HL, Ayala AG, Romsdahl MM. Prognostic factors in chondrosarcoma of bone: a clinicopathologic analysis with emphasis on histologic grading. Cancer 1977;40(2):818‐31. - PubMed
Fiorenza 2002
    1. Fiorenza F, Abudu A, Grimer RJ, Carter SR, Tillman RM, Ayoub K, et al. Risk factors for survival and local control in chondrosarcoma of bone. Journal of Bone and Joint Surgery. British Volume 2002;84(1):93‐9. - PubMed
Fletcher 2013
    1. Fletcher CD, Bridge JA, Hogendoorn P, Mertens F. WHO Classification of Tumours of Soft Tissue and Bone. IARC WHO Classification of Tumours. Fourth. Vol. 5, IARC, 2013.
Geirnaerdt 1997
    1. Geirnaerdt MJ, Hermans J, Bloem JL, Kroon HM, Pope TL, Taminiau AH, et al. Usefulness of radiography in differentiating enchondroma from central grade 1 chondrosarcoma. American Journal of Roentgenology 1997;169(4):1097‐104. - PubMed
Gelderblom 2008
    1. Gelderblom H, Hogendoorn PC, Dijkstra SD, Rijswijk CS, Krol AD, Taminiau AH, et al. The clinical approach towards chondrosarcoma. Oncologist 2008;13(3):320‐9. - PubMed
Giuffrida 2009
    1. Giuffrida AY, Burgueno JE, Koniaris LG, Gutierrez JC, Duncan R, Scully SP. Chondrosarcoma in the United States (1973 to 2003): an analysis of 2890 cases from the SEER database. Journal of Bone and Joint Surgery 2009;91(5):1063‐72. - PubMed
GRADEpro GDT 2015 [Computer program]
    1. McMaster University (developed by Evidence Prime). GRADEpro GDT. Version accessed prior to 30 April 2018. Hamilton (ON): McMaster University (developed by Evidence Prime), 2015.
Healey 1986
    1. Healey JH, Lane JM. Chondrosarcoma. Clinical Orthopaedics and Related Research 1986;204:119‐29. - PubMed
Hickey 2011
    1. Hickey M, Farrokhyar F, Deheshi B, Turcotte R, Ghert M. A systematic review and meta‐analysis of intralesional versus wide resection for intramedullary grade I chondrosarcoma of the extremities. Annals of Surgical Oncology 2011;18(6):1705‐9. - PubMed
Higgins 2003
    1. Higgins JPT, Thompson SG, Deeks JJ, Altman DG. Measuring inconsistency in meta‐analyses. BMJ 2003;327:557‐60. - PMC - PubMed
Jundt 2008
    1. Jundt G, Baumhoer D. Cartilage tumors of the skeleton. Der Pathologe 2008;29(Suppl 2):223‐31. - PubMed
Langendam 2013
    1. Langendam MW, Akl EA, Dahm P, Glasziou P, Guyatt G, Schunemann HJ. Assessing and presenting summaries of evidence in Cochrane Reviews. Systematic Reviews 2013;23(2):81. - PMC - PubMed
Meader 2014
    1. Meader N, King K, Llewellyn A, Norman G, Brown J, Rodgers M, et al. A checklist designed to aid consistency and reproducibility of GRADE assessments: development and pilot validation. Systematic Reviews 2014;3:82. - PMC - PubMed
Mirra 1985
    1. Mirra JM, Gold R, Downs J, Eckardt JJ. A new histologic approach to the differentiation of enchondroma and chondrosarcoma of the bones. A clinicopathologic analysis of 51 cases. Clinical Orthopaedics and Related Research 1985;201:214‐37. - PubMed
Moher 2009
    1. Moher D, Liberati A, Tetzlaff J, Altman DG, The PRISMA Group (2009). Preferred reporting items for systematic reviews and meta‐analyses: The PRISMA Statement. PLoS Medicine 6;7:e1000097. [DOI: 10.1371/journal.pmed1000097] - DOI - PMC - PubMed
Murphey 2003
    1. Murphey MD, Walker EA, Wilson AJ, Kransdorf MJ, Temple HT, Gannon FH. From the archives of the AFIP: imaging of primary chondrosarcoma: radiologic‐pathologic correlation. Radiographics 2003;23(5):1245‐78. - PubMed
Parmar 1998
    1. Parmar MK, Torri V, Stewart L. Extracting summary statistics to perform meta‐analyses of the published literature for survival endpoints. Statistics in Medicine 1998;17(24):2815‐34. - PubMed
Randall 2005
    1. Randall RL, Gowski W. Grade 1 chondrosarcoma of bone: a diagnostic and treatment dilemma. Journal of the National Comprehensive Cancer Network 2005;3(2):149‐56. - PubMed
Review Manager 2014 [Computer program]
    1. Nordic Cochrane Centre, The Cochrane Collaboration. Review Manager 5 (RevMan 5). Version 5.3. Copenhagen: Nordic Cochrane Centre, The Cochrane Collaboration, 2014.
Rosenthal 1984
    1. Rosenthal DI, Schiller AL, Mankin HJ. Chondrosarcoma: correlation of radiological and histological grade. Radiology 1984;150(1):21‐6. - PubMed
Schünemann 2017
    1. Schünemann HJ, Oxman AD, Higgins JP, Vist GE, Glasziou P, Akl E, et al. on behalf of the Cochrane GRADEing Methods Group and the Cochrane Statistical Methods Group. Chapter 11: Completing ‘Summary of findings’ tables and grading the confidence in or quality of the evidence. In: Higgins JPT, Churchill R, Chandler J, Cumpston MS (editors), Cochrane Handbook for Systematic Reviews of Interventions version 5.2.0 (updated June 2017). Cochrane, 2017. Available from www.training.cochrane.org/handbook.
Sterne 2016
    1. Sterne JA, Hernán MA, Reeves BC, Savović J, Berkman ND, Viswanathan M, et al. ROBINS‐I: a tool for assessing risk of bias in non‐randomised studies of interventions. BMJ 2016;355:i4919. - PMC - PubMed
Veth 2005
    1. Veth R, Schreuder B, Beem H, Pruszczynski M. Cryosurgery in aggressive, benign, and low‐grade malignant bone tumours. Lancet Oncology 2005;6(1):25‐34. - PubMed

MeSH terms