Skip to main content
Top
Published in: Annals of Surgical Oncology 12/2021

01-11-2021 | osteosarcoma | Sarcoma

Impact of Secondary Aneurysmal Bone Cysts on Survival of Patients with Enneking Stage IIB Extremity Osteosarcoma: A Propensity Score Matching Analysis

Authors: Chenliang Zhou, MD, Guowei Qian, MD, PhD, Yiyun Wang, MD, Hongtao Li, MD, PhD, Wenxi Yu, MD, Shuier Zheng, MD, PhD, Zan Shen, MD, PhD, Yonggang Wang, MD, PhD

Published in: Annals of Surgical Oncology | Issue 12/2021

Login to get access

Abstract

Background

The clinicopathology of aneurysmal bone cysts (ABCs) secondary to osteosarcoma has not yet been reported. We conduct a retrospective review of ABCs secondary to osteosarcoma to characterize clinicopathology and influence on the survival of patients with Enneking stage IIB extremity osteosarcoma.

Patients and Methods

A total of 300 patients with Enneking stage IIB extremity osteosarcoma were eligible for analysis. These cases were divided, according to the pathology of biopsy and magnetic resonance imaging (MRI), into ABCs group and no ABCs group. Patients (ABCs versus no ABCs) were compared using a 1:2 propensity score analysis to best match between groups. Clinicopathology and survival data were analyzed.

Results

The total occurrence rate of secondary ABCs was 10.3%. A higher prevalence of pathological fractures was observed in the ABCs group (22.6%) compared with the no ABCs group (8.6%) (p = 0.032). Patients with ABCs were more likely to undergo amputation compared with patients without ABCs (p = 0.007). Those with secondary ABCs had poorer response to chemotherapy before and after propensity score matching (p = 0.006 and p = 0.048, respectively). Kaplan–Meier survival analysis showed that EFS and OS distributions were not significantly different between the two patient groups. ABCs were not significantly different in terms of EFS or OS in the multivariate analysis model (p > 0.05).

Conclusions

The presence of secondary ABCs was associated with increased occurrence rate of pathological fracture and high percentage of amputation. Moreover, patients with secondary ABCs had poorer response to chemotherapy. However, the presence of secondary ABCs did not influence survival of patients with Enneking stage IIB extremity osteosarcoma.
Appendix
Available only for authorised users
Literature
1.
go back to reference Casali PG, Bielack S, Abecassis N, et al. Bone sarcomas: ESMO-PaedCan-EURACAN clinical practice guidelines for diagnosis, treatment and follow-up. Ann Oncol. 2018;29(Suppl 4):iv79–95.CrossRef Casali PG, Bielack S, Abecassis N, et al. Bone sarcomas: ESMO-PaedCan-EURACAN clinical practice guidelines for diagnosis, treatment and follow-up. Ann Oncol. 2018;29(Suppl 4):iv79–95.CrossRef
2.
go back to reference Jeon DG, Song WS. How can survival be improved in localized osteosarcoma? Expert Rev Anticancer Ther. 2010;10(8):1313–25.CrossRef Jeon DG, Song WS. How can survival be improved in localized osteosarcoma? Expert Rev Anticancer Ther. 2010;10(8):1313–25.CrossRef
3.
go back to reference Tan PX, Yong BC, Wang J, et al. Analysis of the efficacy and prognosis of limb-salvage surgery for osteosarcoma around the knee. Eur J Surg Oncol. 2012;38(12):1171–7.CrossRef Tan PX, Yong BC, Wang J, et al. Analysis of the efficacy and prognosis of limb-salvage surgery for osteosarcoma around the knee. Eur J Surg Oncol. 2012;38(12):1171–7.CrossRef
4.
go back to reference Rapp TB, Ward JP, Alaia MJ. Aneurysmal bone cyst. J Am Acad Orthop Surg. 2012;20(4):233–41.CrossRef Rapp TB, Ward JP, Alaia MJ. Aneurysmal bone cyst. J Am Acad Orthop Surg. 2012;20(4):233–41.CrossRef
5.
go back to reference Vergel De Dios AM, Bond JR, Shives TC, McLeod RA, Unni KK. Aneurysmal bone cyst A clinicopathologic study of 238 cases. Cancer. 1992;69(12):2921–31.CrossRef Vergel De Dios AM, Bond JR, Shives TC, McLeod RA, Unni KK. Aneurysmal bone cyst A clinicopathologic study of 238 cases. Cancer. 1992;69(12):2921–31.CrossRef
6.
go back to reference Sasaki H, Nagano S, Shimada H, et al. Diagnosing and discriminating between primary and secondary aneurysmal bone cysts. Oncol Lett. 2017;13(4):2290–6.CrossRef Sasaki H, Nagano S, Shimada H, et al. Diagnosing and discriminating between primary and secondary aneurysmal bone cysts. Oncol Lett. 2017;13(4):2290–6.CrossRef
7.
go back to reference Cottalorda J, Kohler R, Sales de Gauzy J, et al. Epidemiology of aneurysmal bone cyst in children: a multicenter study and literature review. J PediatrOrthop B. 2004;13(6):389–94. Cottalorda J, Kohler R, Sales de Gauzy J, et al. Epidemiology of aneurysmal bone cyst in children: a multicenter study and literature review. J PediatrOrthop B. 2004;13(6):389–94.
8.
go back to reference Gutierrez LB, Link TM, Horvai AE, Joseph GB, O’Donnell RJ, Motamedi D. Secondary aneurysmal bone cysts and associated primary lesions: imaging features of 49 cases. Clin Imaging. 2020;62:23–32.CrossRef Gutierrez LB, Link TM, Horvai AE, Joseph GB, O’Donnell RJ, Motamedi D. Secondary aneurysmal bone cysts and associated primary lesions: imaging features of 49 cases. Clin Imaging. 2020;62:23–32.CrossRef
9.
go back to reference Zhao Y, He S, Sun H, et al. Symptomatic aneurysmal bone cysts of the spine: clinical features, surgical outcomes, and prognostic factors. Eur Spine J. 2019;28(6):1537–45.CrossRef Zhao Y, He S, Sun H, et al. Symptomatic aneurysmal bone cysts of the spine: clinical features, surgical outcomes, and prognostic factors. Eur Spine J. 2019;28(6):1537–45.CrossRef
10.
go back to reference Oliveira AM, Perez-Atayde AR, Inwards CY, et al. USP6 and CDH11 Oncogenes identify the neoplastic cell in primary aneurysmal bone cysts and are absent in so-called secondary aneurysmal bone cysts. Am J Pathol. 2004;165(5):1773–80.CrossRef Oliveira AM, Perez-Atayde AR, Inwards CY, et al. USP6 and CDH11 Oncogenes identify the neoplastic cell in primary aneurysmal bone cysts and are absent in so-called secondary aneurysmal bone cysts. Am J Pathol. 2004;165(5):1773–80.CrossRef
11.
go back to reference Jaffe N, Spears R, Eftekhari F, et al. Pathologic fracture in osteosarcoma. Impact of chemotherapy on primary tumor and survival. Cancer. 1987;59(4):701–9.CrossRef Jaffe N, Spears R, Eftekhari F, et al. Pathologic fracture in osteosarcoma. Impact of chemotherapy on primary tumor and survival. Cancer. 1987;59(4):701–9.CrossRef
12.
go back to reference Scully SP, Ghert MA, Zurakowski D, Thompson RC, Gebhardt MC. Pathologic fracture in osteosarcoma: prognostic importance and treatment implications. J Bone Joint Surg Am. 2002;84(1):49–57.CrossRef Scully SP, Ghert MA, Zurakowski D, Thompson RC, Gebhardt MC. Pathologic fracture in osteosarcoma: prognostic importance and treatment implications. J Bone Joint Surg Am. 2002;84(1):49–57.CrossRef
13.
go back to reference Trailescu MD, Pavel AI, Pop MA, et al. Pathological fractures of humerus in children. Therapeutic and pathological considerations. Rom J MorpholEmbryol. 2019;60(3):831–40. Trailescu MD, Pavel AI, Pop MA, et al. Pathological fractures of humerus in children. Therapeutic and pathological considerations. Rom J MorpholEmbryol. 2019;60(3):831–40.
14.
go back to reference Wu Z, Yang X, Xiao J, et al. Aneurysmal bone cyst secondary to giant cell tumor of the mobile spine: a report of 11 cases. Spine (Phila Pa 1976). 2011;36(21):E1385-1390. Wu Z, Yang X, Xiao J, et al. Aneurysmal bone cyst secondary to giant cell tumor of the mobile spine: a report of 11 cases. Spine (Phila Pa 1976). 2011;36(21):E1385-1390.
15.
go back to reference Tang H, Moro A, Feng W, et al. Giant cell tumors combined with secondary aneurysmal bone cysts are more likely to develop postoperative recurrence: a retrospective study of 256 cases. J Surg Oncol. 2019;120(3):359–65.CrossRef Tang H, Moro A, Feng W, et al. Giant cell tumors combined with secondary aneurysmal bone cysts are more likely to develop postoperative recurrence: a retrospective study of 256 cases. J Surg Oncol. 2019;120(3):359–65.CrossRef
16.
go back to reference Anderson ME. Update on survival in osteosarcoma. Orthop Clin North Am. 2016;47(1):283–92.CrossRef Anderson ME. Update on survival in osteosarcoma. Orthop Clin North Am. 2016;47(1):283–92.CrossRef
17.
go back to reference Janeway KA, Barkauskas DA, Krailo MD, et al. Outcome for adolescent and young adult patients with osteosarcoma: a report from the Children’s Oncology Group. Cancer. 2012;118(18):4597–605.CrossRef Janeway KA, Barkauskas DA, Krailo MD, et al. Outcome for adolescent and young adult patients with osteosarcoma: a report from the Children’s Oncology Group. Cancer. 2012;118(18):4597–605.CrossRef
18.
go back to reference Gorlick R, Janeway K, Lessnick S, Randall RL, Marina N, Committee COGBT. Children's Oncology Group's 2013 blueprint for research: bone tumors. Pediatr Blood Cancer. 2013;60(6):1009-1015. Gorlick R, Janeway K, Lessnick S, Randall RL, Marina N, Committee COGBT. Children's Oncology Group's 2013 blueprint for research: bone tumors. Pediatr Blood Cancer. 2013;60(6):1009-1015.
19.
go back to reference Ferrari S, Serra M. An update on chemotherapy for osteosarcoma. Expert Opin Pharmacother. 2015;16(18):2727–36.CrossRef Ferrari S, Serra M. An update on chemotherapy for osteosarcoma. Expert Opin Pharmacother. 2015;16(18):2727–36.CrossRef
20.
go back to reference Traven SA, Brinton DL, Walton ZJ, Leddy LR. A propensity-score matched analysis of limb salvage vs amputation for osteosarcoma. J Surg Oncol. 2019;120(7):1252–8.CrossRef Traven SA, Brinton DL, Walton ZJ, Leddy LR. A propensity-score matched analysis of limb salvage vs amputation for osteosarcoma. J Surg Oncol. 2019;120(7):1252–8.CrossRef
21.
go back to reference Han G, Bi WZ, Xu M, Jia JP, Wang Y. Amputation versus limb-salvage surgery in patients with osteosarcoma: a meta-analysis. World J Surg. 2016;40(8):2016–27.CrossRef Han G, Bi WZ, Xu M, Jia JP, Wang Y. Amputation versus limb-salvage surgery in patients with osteosarcoma: a meta-analysis. World J Surg. 2016;40(8):2016–27.CrossRef
22.
go back to reference He X, Gao Z, Xu H, Zhang Z, Fu P. A meta-analysis of randomized control trials of surgical methods with osteosarcoma outcomes. J Orthop Surg Res. 2017;12(1):5.CrossRef He X, Gao Z, Xu H, Zhang Z, Fu P. A meta-analysis of randomized control trials of surgical methods with osteosarcoma outcomes. J Orthop Surg Res. 2017;12(1):5.CrossRef
23.
go back to reference Jauregui JJ, Nadarajah V, Munn J, et al. Limb salvage versus amputation in conventional appendicular osteosarcoma: a systematic review. Indian J Surg Oncol. 2018;9(2):232–40.CrossRef Jauregui JJ, Nadarajah V, Munn J, et al. Limb salvage versus amputation in conventional appendicular osteosarcoma: a systematic review. Indian J Surg Oncol. 2018;9(2):232–40.CrossRef
24.
go back to reference Kelley LM, Schlegel M, Hecker-Nolting S, et al. Pathological fracture and prognosis of high-grade osteosarcoma of the extremities: an analysis of 2,847 Consecutive Cooperative Osteosarcoma Study Group (COSS) patients. J Clin Oncol. 2020;38(8):823–33.CrossRef Kelley LM, Schlegel M, Hecker-Nolting S, et al. Pathological fracture and prognosis of high-grade osteosarcoma of the extremities: an analysis of 2,847 Consecutive Cooperative Osteosarcoma Study Group (COSS) patients. J Clin Oncol. 2020;38(8):823–33.CrossRef
25.
go back to reference Lewis IJ, Nooij MA, Whelan J, et al. Improvement in histologic response but not survival in osteosarcoma patients treated with intensified chemotherapy: a randomized phase III trial of the European Osteosarcoma Intergroup. J Natl Cancer Inst. 2007;99(2):112–28.CrossRef Lewis IJ, Nooij MA, Whelan J, et al. Improvement in histologic response but not survival in osteosarcoma patients treated with intensified chemotherapy: a randomized phase III trial of the European Osteosarcoma Intergroup. J Natl Cancer Inst. 2007;99(2):112–28.CrossRef
26.
go back to reference Bacci G, Ferrari S, Bertoni F, et al. Long-term outcome for patients with nonmetastatic osteosarcoma of the extremity treated at the istituto ortopedico rizzoli according to the istituto ortopedico rizzoli/osteosarcoma-2 protocol: an updated report. J Clin Oncol. 2000;18(24):4016–27.CrossRef Bacci G, Ferrari S, Bertoni F, et al. Long-term outcome for patients with nonmetastatic osteosarcoma of the extremity treated at the istituto ortopedico rizzoli according to the istituto ortopedico rizzoli/osteosarcoma-2 protocol: an updated report. J Clin Oncol. 2000;18(24):4016–27.CrossRef
27.
go back to reference Bacci G, Ferrari S, Lari S, et al. Osteosarcoma of the limb. Amputation or limb salvage in patients treated by neoadjuvant chemotherapy. J Bone Joint Surg Br. 2002;84(1):88–92.CrossRef Bacci G, Ferrari S, Lari S, et al. Osteosarcoma of the limb. Amputation or limb salvage in patients treated by neoadjuvant chemotherapy. J Bone Joint Surg Br. 2002;84(1):88–92.CrossRef
28.
go back to reference Haynes L, Kaste SC, Ness KK, et al. Pathologic fracture in childhood and adolescent osteosarcoma: a single-institution experience. Pediatr Blood Cancer. 2017;64(4):e26290.CrossRef Haynes L, Kaste SC, Ness KK, et al. Pathologic fracture in childhood and adolescent osteosarcoma: a single-institution experience. Pediatr Blood Cancer. 2017;64(4):e26290.CrossRef
Metadata
Title
Impact of Secondary Aneurysmal Bone Cysts on Survival of Patients with Enneking Stage IIB Extremity Osteosarcoma: A Propensity Score Matching Analysis
Authors
Chenliang Zhou, MD
Guowei Qian, MD, PhD
Yiyun Wang, MD
Hongtao Li, MD, PhD
Wenxi Yu, MD
Shuier Zheng, MD, PhD
Zan Shen, MD, PhD
Yonggang Wang, MD, PhD
Publication date
01-11-2021
Publisher
Springer International Publishing
Published in
Annals of Surgical Oncology / Issue 12/2021
Print ISSN: 1068-9265
Electronic ISSN: 1534-4681
DOI
https://doi.org/10.1245/s10434-021-09878-z

Other articles of this Issue 12/2021

Annals of Surgical Oncology 12/2021 Go to the issue