Proton beam therapy for a patient with large rhabdomyosarcoma of the body trunk
© Takizawa et al. 2015
Received: 2 July 2015
Accepted: 8 November 2015
Published: 16 November 2015
We present the clinical course of a pediatric patient with large rhabdomyosarcoma of the body trunk who received proton beam therapy (PBT).
A 1-year-old girl was diagnosed with stage IV alveolar rhabdomyosarcoma in 2008. A large tumor was located in the central diaphragm and had infiltrated the liver and pericardium with peritoneal dissemination. Chemotherapy was immediately started with six courses of vincristine, actinomycin-D and cyclophosphamide (VAC) firstly, and secondly followed by 2 courses of ifosfamide, carboplatin and etoposide (ICE), but a large tumor of 15 cm in size remained. The tumor was inoperable because of its location, and photon radiotherapy could not be performed due to limited liver tolerance. The patient was referred to our hospital and received PBT at a dose of 54 GyE in 30 fractions in June 2009.
The tumor quickly responded and 95 % of volume reduction was achieved at the end of PBT. However, marginal recurrence in the caudal part of the irradiated field, where we reduced the proton dose because of the presence of the intestine, was detected in August 2010. The recurrent tumor size was less than 1 cm. Chemotherapy with VAC followed by topotecan and carboplatin (TC) was again tried, but the tumor size was stable. Repeated PBT was not possible because of limited intestinal tolerance; therefore, intraoperative radiotherapy was conducted with 20 Gy of electron beams in April 2011. The tumor was subsequently well controlled, but secondary myelodysplastic syndrome developed and the patient died of hemophagocytic syndrome after umbilical cord blood transplantation in May 2012.
PBT was performed safely and effectively for a 1-year-old girl with alveolar rhabdomyosarcoma with liver and cardiac invasion that was resistant to surgery and chemotherapy. This case illustrates that PBT can be useful in cases that are difficult to treat with conventional radiotherapy.
Radiotherapy plays an important role in treatment of rhabdomyosarcoma (RMS), with a dose of 40–60 Gy typically required, and hyperfractionation (1.1-Gy twice daily fractions) or standard fractionation (1.8-Gy daily fractions) is used for definitive treatment . The risk of morbidity radiation-related should be carefully considered depending on the volume and the dose delivered in the pediatric patients. We experienced a pediatric patient with an unresectable large alveolar RMS with liver invasion. Photon radiotherapy was considered as the first treatment choice, but was not administered because of the lack of tolerance of the liver. Proton beam therapy (PBT) was chosen in this case, since PBT is a particle radiotherapy with excellent dose localization because of the sharp and narrow Bragg peak [2, 3]. Here, we present the clinical course of the patient after treatment with PBT.
In this case, PBT was performed safely and effectively with alveolar rhabdomyosarcoma with liver and cardiac invasion that was unable to be removed with surgery and was resistant to chemotherapy. We supposed that side effect and tumor’s local control turned worse, if we used conventional radiotherapy not PBT. This case illustrates that PBT can be useful in cases that are difficult to treat with conventional radiotherapy.
Written informed consent was obtained from the patient’s legal guardians for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
This work was supported in part by Grants-in-Aid for Scientific Research (B) (24390286); Challenging Exploratory Research (24659556), Young Scientists (B) (25861064); and Scientific Research (C) (24591832) from the Ministry of Education, Science, Sports and Culture of Japan. We wish to thank the members of the Tsukuba Critical Path Research and Education Integrated Leading Center (CREIL) at the University of Tsukuba for their critical advice in conducting the study and data management during the study period. This work was presented at a conference of the International Society of Pediatric Oncology 2013, Hong Kong (Pediatric Blood Cancer. 2013;60(S3):93.).
Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
- Donaldson SS, Meza J, Breneman JC, Crist WM, Laurie F, Qualman SJ, et al. Results from the IRS-IV randomized trial of hyperfractionated radiotherapy in children with rhabdomyosarcoma-a report from the IRSG. Int J Radiat Oncol Biol Phys. 2001;51:718–28.View ArticlePubMedGoogle Scholar
- Oshiro Y, Okumura T, Mizumoto M, Fukushima T, Ishikawa H, Hashimoto T, et al. Proton beam therapy for unresectable hepatoblastoma in children: survival in one case. Acta Oncol. 2013;52:600–3.View ArticlePubMedGoogle Scholar
- Mizumoto M, Tokuuye K, Sugahara S, Nakayama H, Fukumitsu N, Ohara K. Proton beam therapy for hepatocellular carcinoma adjacent to the porta hepatis. Int J Radiat Oncol Biol Phys. 2008;71:462–7.View ArticlePubMedGoogle Scholar
- Kanemoto A, Hirayama R, Moritake T, Furusawa Y, Sun L, Sakae T, et al. RBE and OER within the spread-out Bragg peak for proton beam therapy: in vitro study at the Proton Medical Research Center at the University of Tsukuba. J Radiat Res. 2014;55:1028–32.PubMed CentralView ArticlePubMedGoogle Scholar
- Ries LAG, Smith MA, Gurney JG, Linet M, Tamra T, Young JL, Bunin GR (eds). Cancer Incidence and Survival among Children and Adolescents: United States SEER Program 1975–1995, National Cancer Institute, SEER Program. NIH Pub. No. 99–4649. Bethesda, MD; 1999.Google Scholar
- McDowell HP. Update on childhood rhabdomyosarcoma. Arch Dis Child. 2003;88:354–7.PubMed CentralView ArticlePubMedGoogle Scholar
- Breneman JC, Lyden E, Pappo AS, Link MP, Anderson JR, Parham DM, et al. Prognostic factors and clinical outcomes in children and adolescents with metastatic rhabdomyosarcoma: a report from the intergroup rhabdomyosarcoma study IV. J Clin Oncol. 2003;21:78–84.View ArticlePubMedGoogle Scholar
- Crist WM, Anderson JR, Meza JL, Fryer C, Raney RB, Ruymann FB, et al. Intergroup rhabdomyosarcoma study-IV: results for patients with nonmetastatic disease. J Clin Oncol. 2001;19:3091–102.PubMedGoogle Scholar
- Wolden SL, Anderson JR, Crist WM, Breneman JC, Wharam Jr MD, Wiener ES, et al. Indications for radiotherapy and chemotherapy after complete resection in rhabdomyosarcoma: a report from the intergroup rhabdomyosarcoma studies I to III. J Clin Oncol. 1999;17:3468–75.PubMedGoogle Scholar
- Regine WF, Fontanesi J, Kumar P, Ayers D, Bowman LC, Pappo AS, et al. Local tumor control in rhabdomyosarcoma following low-dose irradiation: Comparison of group II and select group III patients. Int J Radiat Oncol Biol Phys. 1995;31:485–91.View ArticlePubMedGoogle Scholar
- Emami B, Lyman J, Brown A, Coia L, Goitein M, Munzenrider JE, et al. Tolerance of normal tissue to therapeutic irradiation. Int J Radiat Oncol Biol Phys. 1991;21:109–22.View ArticlePubMedGoogle Scholar
- Kanemoto A, Mizumoto M, Okumura T, Takahashi H, Hashimoto T, Oshiro Y, et al. Dose-volume histogram analysis for risk factors of radiation-induced rib fracture after hypofractionated proton beam therapy for hepatocellular carcinoma. Acta Oncol. 2013;52:538–44.View ArticlePubMedGoogle Scholar
- Mizumoto M, Okumura T, Hashimoto T, Fukuda K, Oshiro Y, Fukumitsu N, et al. Proton beam therapy for hepatocellular carcinoma: a comparison of three treatment protocols. Int J Radiat Oncol Biol Phys. 2011;81:1039–45.View ArticlePubMedGoogle Scholar
- Mizumoto M, Oshiro Y, Ayuzawa K, Miyamoto T, Okumura T, Fukushima T, et al. Preparation of pediatric patients for treatment with proton beam therapy. Radiother Oncol. 2015;114:245–8.View ArticlePubMedGoogle Scholar
- Cotter SE, Herrup DA, Friedmann A, Macdonald SM, Pieretti RV, Robinson G, et al. Proton radiotherapy for pediatric bladder/prostate rhabdomyosarcoma: clinical outcomes and dosimetry compared to intensity-modulated radiation therapy. Int J Radiat Oncol Biol Phys. 2011;81:1367–73.View ArticlePubMedGoogle Scholar
- Timmermann B, Schuck A, Niggli F, Weiss M, Lomax AJ, Pedroni E, et al. Spot-scanning proton therapy for malignant soft tissue tumors in childhood: First experiences at the Paul Scherrer Institute. Int J Radiat Oncol Biol Phys. 2007;67:497–504.View ArticlePubMedGoogle Scholar
- Ladra MM, Szymonifka JD, Mahajan A, Friedmann AM, Yong Yeap B, Goebel CP, et al. Preliminary results of a phase II trial of proton radiotherapy for pediatric rhabdomyosarcoma. J Clin Oncol. 2014;32:3762–70.View ArticlePubMedGoogle Scholar
- Tsunashima Y, Sakae T, Shioyama Y, Kagei K, Terunuma T, Akihiro N, et al. Correlation between the respiratory waveform measured using a respiratory sensor and 3D tumor motion in gated radiotherapy. Int J Radiat Oncol Biol Phys. 2004;60(3):951–8. 11.View ArticlePubMedGoogle Scholar