Abstract
Background
Outcomes in adult patients with rhabdomyosarcoma are poor, with a 5-year survival rate of approximately 30 %. The current study aimed to compare the clinical outcomes of adult and childhood rhabdomyosarcoma patients with local and metastatic disease and to examine the impact and timing of local therapy on metastasis.
Methods
Clinicopathological features and patient outcomes were reviewed retrospectively for rhabdomyosarcoma patients receiving chemotherapy between 1981 and 2010 at our institution. Adults were defined as those aged 21 years or older.
Results
Of the 98 patients identified, 36 were adults (median age, 29; range, 21–72) and 62 were children (median age, 11; range, 0.6–20). Median progression-free survival of localized and metastatic disease for children and adults was as follows: localized disease, 166.9 versus 22.4 months (p = 0.005), and metastatic disease, 13.3 versus 13.3 months (p = 0.949), respectively. Multivariate regression analysis revealed that older age (≥21 vs. <21) was a significant poor prognostic factor in localized disease. Conversely, age was not related to survival in metastatic disease. Receiving radiotherapy to the primary site was an independent factor indicating a better prognosis. An analysis of the optimal timing of local therapy was performed for 53 patients; however, its significance on survival could not be determined.
Conclusions
Age was a negative prognostic factor in rhabdomyosarcoma patients with localized disease, but it did not affect the survival in metastatic disease. For metastatic disease, although local therapies may be effective for survival, the timing of such therapies should be determined individually.
Similar content being viewed by others
References
Arndt CA, Stoner JA, Hawkins DS, Rodeberg DA, Hayes-Jordan AA, Paidas CN, Parham DM, Teot LA, Wharam MD, Breneman JC, Donaldson SS, Anderson JR, Meyer WH (2009) Vincristine, actinomycin, and cyclophosphamide compared with vincristine, actinomycin, and cyclophosphamide alternating with vincristine, topotecan, and cyclophosphamide for intermediate-risk rhabdomyosarcoma: children’s oncology group study D9803. J Clin Oncol 27(31):5182–5188. doi:10.1200/JCO.2009.22.3768
Breitfeld PP, Meyer WH (2005) Rhabdomyosarcoma: new windows of opportunity. Oncologist 10(7):518–527. doi:10.1634/theoncologist.10-7-518
Breneman JC, Lyden E, Pappo AS, Link MP, Anderson JR, Parham DM, Qualman SJ, Wharam MD, Donaldson SS, Maurer HM, Meyer WH, Baker KS, Paidas CN, Crist WM (2003) Prognostic factors and clinical outcomes in children and adolescents with metastatic rhabdomyosarcoma–a report from the intergroup rhabdomyosarcoma study IV. J Clin Oncol 21(1):78–84
Carli M, Colombatti R, Oberlin O, Bisogno G, Treuner J, Koscielniak E, Tridello G, Garaventa A, Pinkerton R, Stevens M (2004) European intergroup studies (MMT4-89 and MMT4-91) on childhood metastatic rhabdomyosarcoma: final results and analysis of prognostic factors. J Clin Oncol 22(23):4787–4794. doi:10.1200/JCO.2004.04.083
Crist W, Gehan EA, Ragab AH, Dickman PS, Donaldson SS, Fryer C, Hammond D, Hays DM, Herrmann J, Heyn R et al (1995) The third intergroup rhabdomyosarcoma study. J Clin Oncol 13(3):610–630
Crist WM, Anderson JR, Meza JL, Fryer C, Raney RB, Ruymann FB, Breneman J, Qualman SJ, Wiener E, Wharam M, Lobe T, Webber B, Maurer HM, Donaldson SS (2001) Intergroup rhabdomyosarcoma study-IV: results for patients with nonmetastatic disease. J Clin Oncol 19(12):3091–3102
Esnaola NF, Rubin BP, Baldini EH, Vasudevan N, Demetri GD, Fletcher CD, Singer S (2001) Response to chemotherapy and predictors of survival in adult rhabdomyosarcoma. Ann Surg 234(2):215–223
Ferrari A, Dileo P, Casanova M, Bertulli R, Meazza C, Gandola L, Navarria P, Collini P, Gronchi A, Olmi P, Fossati-Bellani F, Casali PG (2003) Rhabdomyosarcoma in adults. A retrospective analysis of 171 patients treated at a single institution. Cancer 98(3):571–580. doi:10.1002/cncr.11550
Hawkins WG, Hoos A, Antonescu CR, Urist MJ, Leung DH, Gold JS, Woodruff JM, Lewis JJ, Brennan MF (2001) Clinicopathologic analysis of patients with adult rhabdomyosarcoma. Cancer 91(4):794–803. doi:10.1002/1097-0142(20010215)91:4<794:AID-CNCR1066>3.0.CO;2-Q
Klingebiel T, Boos J, Beske F, Hallmen E, Int-Veen C, Dantonello T, Treuner J, Gadner H, Marky I, Kazanowska B, Koscielniak E (2008) Treatment of children with metastatic soft tissue sarcoma with oral maintenance compared to high dose chemotherapy: report of the HD CWS-96 trial. Pediatr Blood Cancer 50(4):739–745. doi:10.1002/pbc.21494
La Quaglia MP, Heller G, Ghavimi F, Casper ES, Vlamis V, Hajdu S, Brennan MF (1994) The effect of age at diagnosis on outcome in rhabdomyosarcoma. Cancer 73(1):109–117
La TH, Wolden SL, Su Z, Linardic C, Randall RL, Hawkins DS, Donaldson SS (2011) Local therapy for rhabdomyosarcoma of the hands and feet: is amputation necessary? A report from the Children’s Oncology Group. Int J Radiat Oncol Biol Phys 80(1):206–212. doi:10.1016/j.ijrobp.2010.01.053
Lager JJ, Lyden ER, Anderson JR, Pappo AS, Meyer WH, Breitfeld PP (2006) Pooled analysis of phase II window studies in children with contemporary high-risk metastatic rhabdomyosarcoma: a report from the Soft Tissue Sarcoma Committee of the Children’s Oncology Group. J Clin Oncol 24(21):3415–3422. doi:10.1200/JCO.2005.01.9497
Leaphart C, Rodeberg D (2007) Pediatric surgical oncology: management of rhabdomyosarcoma. Surg Oncol 16(3):173–185. doi:10.1016/j.suronc.2007.07.003
Little DJ, Ballo MT, Zagars GK, Pisters PW, Patel SR, El-Naggar AK, Garden AS, Benjamin RS (2002) Adult rhabdomyosarcoma: outcome following multimodality treatment. Cancer 95(2):377–388. doi:10.1002/cncr.10669
Maurer HM, Beltangady M, Gehan EA, Crist W, Hammond D, Hays DM, Heyn R, Lawrence W, Newton W, Ortega J et al (1988) The intergroup rhabdomyosarcoma study-I. A final report. Cancer 61(2):209–220
Maurer HM, Gehan EA, Beltangady M, Crist W, Dickman PS, Donaldson SS, Fryer C, Hammond D, Hays DM, Herrmann J et al (1993) The Intergroup Rhabdomyosarcoma Study-II. Cancer 71(5):1904–1922
Meza JL, Anderson J, Pappo AS, Meyer WH (2006) Analysis of prognostic factors in patients with nonmetastatic rhabdomyosarcoma treated on intergroup rhabdomyosarcoma studies III and IV: the Children’s Oncology Group. J Clin Oncol 24(24):3844–3851. doi:10.1200/JCO.2005.05.3801
Michalski JM, Meza J, Breneman JC, Wolden SL, Laurie F, Jodoin M, Raney B, Wharam MD, Donaldson SS (2004) Influence of radiation therapy parameters on outcome in children treated with radiation therapy for localized parameningeal rhabdomyosarcoma in Intergroup Rhabdomyosarcoma Study Group trials II through IV. Int J Radiat Oncol Biol Phys 59(4):1027–1038. doi:10.1016/j.ijrobp.2004.02.064
Minn AY, Lyden ER, Anderson JR, Million L, Arndt CA, Brown K, Hawkins DS, Donaldson SS (2010) Early treatment failure in intermediate-risk rhabdomyosarcoma: results from IRS-IV and D9803–a report from the Children’s Oncology Group. J Clin Oncol 28(27):4228–4232. doi:10.1200/JCO.2010.29.0247
Oberlin O, Rey A, Lyden E, Bisogno G, Stevens MC, Meyer WH, Carli M, Anderson JR (2008) Prognostic factors in metastatic rhabdomyosarcomas: results of a pooled analysis from United States and European cooperative groups. J Clin Oncol 26(14):2384–2389. doi:10.1200/JCO.2007.14.7207
Pappo AS, Shapiro DN, Crist WM (1997) Rhabdomyosarcoma. Biology and treatment. Pediatr Clin North Am 44(4):953–972
Pappo AS, Lyden E, Breitfeld P, Donaldson SS, Wiener E, Parham D, Crews KR, Houghton P, Meyer WH (2007) Two consecutive phase II window trials of irinotecan alone or in combination with vincristine for the treatment of metastatic rhabdomyosarcoma: the Children’s Oncology Group. J Clin Oncol 25(4):362–369. doi:10.1200/JCO.2006.07.1720
Raney RB, Maurer HM, Anderson JR, Andrassy RJ, Donaldson SS, Qualman SJ, Wharam MD, Wiener ES, Crist WM (2001) The Intergroup Rhabdomyosarcoma Study Group (IRSG): major lessons from the IRS-I through IRS-iv studies as background for the current IRS-V treatment protocols. Sarcoma 5(1):9–15. doi:10.1080/13577140120048890
Raney RB, Meza J, Anderson JR, Fryer CJ, Donaldson SS, Breneman JC, Fitzgerald TJ, Gehan EA, Michalski JM, Ortega JA, Qualman SJ, Sandler E, Wharam MD, Wiener ES, Maurer HM, Crist WM (2002) Treatment of children and adolescents with localized parameningeal sarcoma: experience of the Intergroup Rhabdomyosarcoma Study Group protocols IRS-II through -IV, 1978–1997. Med Pediatr Oncol 38(1):22–32. doi:10.1002/mpo.1259
Rodeberg DA, Anderson JR, Arndt CA, Ferrer FA, Raney RB, Jenney ME, Brecht IB, Koscielniak E, Carli M, Bisogno G, Oberlin O, Rey A, Ullrich F, Stevens MC, Meyer WH (2011) Comparison of outcomes based on treatment algorithms for rhabdomyosarcoma of the bladder/prostate: combined results from the Children’s Oncology Group, German Cooperative Soft Tissue Sarcoma Study, Italian Cooperative Group, and International Society of Pediatric Oncology Malignant Mesenchymal Tumors Committee. Int J Cancer 128(5):1232–1239. doi:10.1002/ijc.25444
Schuck A, Mattke AC, Schmidt B, Kunz DS, Harms D, Knietig R, Treuner J, Koscielniak E (2004) Group II rhabdomyosarcoma and rhabdomyosarcoma like tumors: is radiotherapy necessary? J Clin Oncol 22(1):143–149. doi:10.1200/JCO.2004.04.180
Sultan I, Qaddoumi I, Yaser S, Rodriguez-Galindo C, Ferrari A (2009) Comparing adult and pediatric rhabdomyosarcoma in the surveillance, epidemiology and end results program, 1973 to 2005: an analysis of 2,600 patients. J Clin Oncol 27(20):3391–3397. doi:10.1200/JCO.2008.19.7483
Conflict of interest
None.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Kojima, Y., Hashimoto, K., Ando, M. et al. Clinical outcomes of adult and childhood rhabdomyosarcoma treated with vincristine, d-actinomycin, and cyclophosphamide chemotherapy. J Cancer Res Clin Oncol 138, 1249–1257 (2012). https://doi.org/10.1007/s00432-012-1199-x
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00432-012-1199-x