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PubMed2004

Acetabular osteoarticular allograft after Ewing's sarcoma resection.

Ewing's sarcoma of the pelvis traditionally has been a difficult problem in orthopaedic oncology. Multiple studies have had inferior outcomes when compared with outcomes of patients with similar stage disease involving only the extremities. A major reason for this discrepancy is the difficulty associated with resection and reconstruction of pelvic tumors. Often the tumors are deemed nonresectable and are treated with chemotherapy and radiation or radiation alone. Patients treated in this manner have lower 5-year survival rates, higher local recurrence rates, and multiple long-term complications. The case of a 4-year-old girl with Ewing's sarcoma of the left hemi-pelvis is reported. Combined with adjuvant chemotherapy, this patient was treated with resection followed by osteoarticular allograft reconstruction. At 2 years followup, the patient was disease-free with an excellent functional outcome.

Acetabulum↗
PubMed1996

Significance of surgical margin on the prognosis of patients with Ewing's sarcoma. A report from the Cooperative Ewing's Sarcoma Study.

BACKGROUND: There is little information regarding an adequate surgical margin for local control of Ewing's sarcoma. METHODS: Two hundred and forty-four patients (PTS) with Ewing's sarcoma who were registered in the Cooperative Ewing's Sarcoma Studies underwent surgical treatment. Ninety-four PTS underwent definitive surgery (surgery alone), 131 PTS received postoperative irradiation, and 19 PTS received preoperative irradiation. The surgical margins were distributed as follows: radical, 29 PTS; wide, 148 PTS; marginal, 39 PTS; and intralesional, 28 PTS. The impact of the surgical margin on the treatment outcome of PTS was analyzed statistically. RESULTS: The local or combined (local recurrence and systemic metastasis) relapse rate after surgery with or without irradiation was significantly lower compared with that after definitive irradiation (irradiation alone) (7% vs. 31%, P < 0.0001). The local or combined relapse rate after complete resection (radical or wide margin) with or without irradiation was less compared with that after incomplete resection (marginal or intralesional margin) with or without irradiation (5% vs. 12% P = 0.0455). The local or combined relapse rate did not greatly decreased after irradiation after incomplete surgery (from 14% to 12%). In both groups of good (viable tumor cells < 10%) and poor (viable cells > or = 10%) histologic response, the difference in systemic or combined relapse rate between patients undergoing complete and incomplete surgery was not significant. The 10-year overall survival of the PTS for each of the margins was distributed as follows: radical, 58%; wide, 65%; marginal, 61%; and intralesional, 71% (P = not significant). CONCLUSIONS: Surgery in patients with Ewing's sarcoma adds to the safety of local control. Under the current treatment regimen with intensive chemotherapy and irradiation, complete resection of the tumor appears capable of decreasing the risk of local recurrence.

Adolescent↗
PubMed1988

Radio-induced sarcomas in survivors of Ewing's sarcoma.

Of 255 cases of Ewing's sarcoma recorded at the Bone Tumor Center of the Rizzoli Orthopaedic Institute, 78 patients (irradiated and with a follow-up of longer than 3 years) were considered "at risk" for the development of a second radio-induced sarcoma (RIS). Three of the 78 patients developed an RIS in the irradiated field. Theoretical and statistical analyses were carried out considering different modalities of local treatment. Statistically, the only significant factor was related to the irradiation dose. Surgical resection seems to prevent RIS.

Adolescent↗
PubMed2003

Potentials for RNAi in sarcoma research and therapy: Ewing's sarcoma as a model.

Existing data identify EWS-FLI1 as indispensable for sustained Ewing's sarcoma growth and as the ideal therapeutic target in this disease. The siRNA may hold great promises as a fusion gene specific agent. RNAi mediated suppression of EWS-FLI1 is likely to result in an altered tumor cell phenotype including changes in chemosensitivity, and a restored differentiation potential. Thus, RNAi may serve as an adjuvant to chemotherapy. As a therapeutic means however, RNAi is hampered by limitations in the delivery of the agent and emergence of resistant clones. In vitro suppression of EWS-FLI1 expression will allow to define the phenotypic characteristics of dormant tumor cells that may give rise to late relapses, enabling improved diagnosis and treatment even of minimal residual disease.

Animals↗
PubMed1981

Radiation therapy in the multimodal management of Ewing's sarcoma of bone: report of the Intergroup Ewing's Sarcoma Study.

This paper is a progress report on the role of radiation therapy (RT) in local tumor control and the decreased incidence of pulmonary metastasis in 251 patients entered in the Intergroup Ewing's Sarcoma Study. All were followed for more that 1 year, and their RT records were reviewed. Doses to the primary tumor in the range of 4,500--6,500 rad were administered over approximately 5 to 6 weeks in combination with 4 drugs, i.e., vincristine (VCR), dactinomycin (DAC), cyclophosphamide (CY), and adriamycin, or only the first 3. One group of patients received the 3 drugs and bilateral pulmonary irradiation (approximately 1,500 rad in 2 wk). Preliminary analysis showed a local primary tumor control of approximately 90%. Patients with lesions in the pelvis and humerus had local failure rates of 13% (7 of 54) and 21.4% (6 of 28), respectively. The treatment groups differed significantly in the incidence of pulmonary metastasis. Patients treated with the 4 drugs (regimen 1) had a 14% incidence, whereas 42% of those treated with only 3 drugs (regimen 2) developed pulmonary metastases. Of all patients treated with 3 drugs and pulmonary irradiation (regimen 3), 18% showed lung metastases. The study indicated that intensive chemotherapy and RT significantly improved the local control and survival of patients with localized Ewing's sarcoma. However, the high incidence of metastasis indicated the need for more effective systemic chemotherapy for further improvement of treatment results. More studies are needed so we can define the volume to be treated and the optimal dose of irradiation to determine a therapeutic strategy that will yield optimal survival and tumor control with the fewest sequelae.

Adolescent↗
PubMed1995

Ewing's sarcoma of the ribs. A report from the cooperative Ewing's sarcoma study.

31 patients with primary Ewing's sarcoma of the ribs were treated according to the protocols of CESS 81, CESS 86P and CESS 86. The results of treatment were reviewed and analysed. 24 patients presented with localised disease and 7 with regional disease. 20 of 24 localised cases and 6 of 7 regional cases underwent tumour resection. All but 2 localised cases received irradiation. The cumulative relapse-free survival (RFS) rate of 31 patients was 61% at 12.8 years. Patients with poor prognosis had tumour of the upper ribs (P = 0.0338), the posterior component of the ribs (P = 0.0597), or regional disease (P = 0.0001). Tumour size, existence of pleural effusion, type of the surgical margin and response to chemotherapy were not significant prognostic factors. Most of the localised cases could be controlled by combined treatment, but in regional cases prognosis remained poor.

Adolescent↗
PubMed1982

Intramyofiber skeletal muscle invasion in Ewing's sarcoma of bone: clinicopathologic observations from the intergroup Ewing's sarcoma study.

Skeletal muscle may be infiltrated by a variety of invasive or metastatic malignant tumors. In 14 out of 291 cases of Ewing's sarcoma, we observed diagnostic biopsies in which local skeletal muscle fibers contained intrasarcolemmal aggregates of tumor cells. Cases demonstrating local intramyofiber invasion have a higher mitotic count, an increased rate of developing distant metastases, and a decreased survival when compared to cases not demonstrating this skeletal muscle "parasitism." The usefulness of skeletal muscle intramyofiber invasion in the differential diagnosis of Ewing's sarcoma remains unknown at this time.

Biopsy↗
PubMed1983

Primary Ewing's sarcoma of the ribs. A report from the intergroup Ewing's sarcoma study.

Thirty-six patients with primary Ewing's sarcoma of the ribs have been reviewed. Of these, 21 had clinically localized disease at diagnosis and were entered on protocol IESS 7299, eight had regional and seven metastatic disease at diagnosis and were entered on protocol 7450. The 21 with localized disease were treated with surgical excision or biopsy, followed by local radiotherapy (in all but one patient) and randomization to one of three chemotherapy regimens. Eleven patients (52%) remain disease-free for periods ranging from 18 to 64 months, respectively. Seven of eight patients who underwent complete surgical excision of the primary lesion remain disease-free, compared with four of 12 (excluding one patient who died disease-free) who remain disease-free after partial excision or biopsy. However, analysis of size of tumor at diagnosis reveals that smaller primary tumors have a better prognosis irrespective of extent of surgery. Protocol IESS 7450, consisted of radiotherapy to all areas of known disease and four drug chemotherapy. Four (50%) with regional disease but none with metastatic disease have remained alive and continuously disease-free. It is concluded that an aggressive approach to Ewing's sarcoma of the ribs is justified by the results, as even regional disease may be curable. The apparent prognostic advantage for those patients undergoing surgical excision may be explained by patient selection.

Adolescent↗
PubMed1952

Ewing's sarcoma.

Explore the source record for details and available documents.

Humans↗
PubMed2005

A multicenter Phase II study of bortezomib in recurrent or metastatic sarcomas.

BACKGROUND: Based on evidence of activity in preclinical and Phase I studies, the authors undertook a study of bortezomib, a reversible proteasome inhibitor, for patients with metastatic sarcomas. METHODS: Two arms were opened, each using a Simon two-stage design. Arm A included patients with osteogenic sarcoma, Ewing sarcoma, and rhabdomyosarcoma. Arm B accrued patients with other types of soft tissue sarcomas. Patients were not allowed to have received previous chemotherapy for metastatic disease. The initial dose of bortezomib was a 1.5 mg/m2 intravenous push twice weekly followed by a rest week. The dose was escalated to 1.7 mg/m2 if patients tolerated Cycle 1 well. The dose escalation was eliminated due to toxicity observed in the first six patients. RESULTS: Painful neuropathy, myalgias, and asthenia were the most significant observed toxicities. The most frequent toxicities included fatigue, diarrhea, constipation, and nausea. Pharmacodynamic data from 18 patients with complete data collection did not show consistent differences between patients with or without Grade 2 or Grade 3 neuropathy (toxicity graded according the National Cancer Institute Common Toxicity Criteria). Arm A had low accrual and was closed. One confirmed partial response among 21 evaluable patients was observed on Arm B in a patient with leiomyosarcoma. Due to the inactivity of this agent, the study was closed after the first stage of accrual. CONCLUSIONS: Bortezomib has minimal activity in soft tissue sarcoma as a single agent. If studied further in sarcomas, bortezomib should be investigated in combination with agents with demonstrated preclinical synergy.

Adult↗
PubMed1989

[Treatment of otherwise incurable tumor diseases in childhood using whole-body hyperthermia and chemotherapy].

Conventional methods of treatment having failed in 17 children (aged 9/12 to 16 5/12 years) with incurable solid malignant tumours underwent whole-body hyperthermia (41.8-42.0 degrees C, for 2-3 h), hyperglycaemia (20-25 mmol/l) and polychemotherapy. Five children had neuroblastoma (stage 4), three Wilm's tumour (stage 4 or 5, unfavourable histology), five skeletal sarcoma with metastases, three inoperable malignant liver tumour, and one brainstem tumour of unknown histology. Whole-body hyperthermia was induced by extracorporeal blood warming in an haemodialysis apparatus under neuroleptic analgesia, thermistors measuring the temperature in the oesophagus, rectum, trachea and skin. There were on average four treatment sessions (between one and ten, total 58), a week apart. The result could be assessed in 12 children: one persisting complete remission (19 months-metastasising renal rhabdoid tumour), eight partial or incomplete remissions, and three nonresponders (osteogenic sarcoma; Ewing sarcoma; brainstem tumour). If the risk can be satisfactorily judged the method is useful and of bearable toxicity. The results point to a high antitumour effectiveness of combined hyperthermia and chemotherapy.

Adolescent↗
PubMed2005

Overview of sarcomas in the adolescent and young adult population.

Based on the data of the Surveillance, Epidemiology and End Results Section of the National Cancer Institute (SEER) program, soft tissue and bone sarcomas account for about 1% of all new malignancies diagnosed in the United States each year. However, there are numerous different histologic types, and any given type of sarcoma is extremely rare. Determining the incidence of sarcomas by age and type is difficult due to the limited data reported. The SEER program collects data regarding age but only limited data on histology, while most series reported in the literature include either adults or pediatric patients, but rarely both. In an effort to estimate the frequency and absolute numbers of different sarcomas in the adolescent and young adult population, the University of Texas M. D. Anderson Cancer Center (MDACC) tumor registry was queried for all soft tissue sarcomas from 1990 through 2003, and all bone sarcomas from 1990 through 2002. Based on this query, an overview of sarcomas that occur predominantly in the adolescent and young adult (AYA) population is presented. These sarcomas include rhabdomyosarcoma, synovial sarcoma, neurogenic sarcoma, epithelioid sarcomas, alveolar soft parts sarcoma, Ewing sarcoma, and osteosarcoma. Using the percentages for occurrence of each histologic type determined from the MDACC database, and the SEER estimate of overall sarcoma incidence, an estimate of the number of new cases in 2004 for the predominant histologic types occurring in the AYA population are presented. Also reviewed are the chromosomal translocations that occur frequently in sarcomas presenting in the AYA population.

Adolescent↗
PubMed2002

Fractionated perioperative high dose rate brachytherapy using a tissue equivalent bendy applicator.

Intraoperative radiation techniques allow an additional local dose in areas at high-risk for local failure. With brachytherapy techniques, perioperative radiation can be fractionated. Fractionated treatment might offer an interesting alternative to a single dose, both to increase the therapeutic ratio and to protect late reacting tissues at risk. The dose distribution for brachytherapy applicators can be optimized using spacer materials. In this prospective study a new tissue equivalent bendy applicator (TEBA) that can remain in situ for several days is introduced, and the feasibility of fractionated perioperative high dose rate (HDR) brachytherapy is examined. 31 patients with different tumours (soft tissue sarcoma, Ewings sarcoma, rectal cancer, and locally infiltrating diseases) were treated. The TEBA was applied, depending on resection status and intraoperative findings. Planning was based on digitized radiographs and CT scans. Perioperative HDR brachytherapy was performed using an individual treatment schedule. In 29 patients perioperative radiation was given and in 26 cases fractionated brachytherapy application was possible. TEBA application time varied from 1 day to 11 days. During this time between 1 and 8 fractions were given with total doses from 10 Gy to 25 Gy. Fractionated perioperative brachytherapy with this technique is feasible and adequate. Further studies will show whether fractionated perioperative treatment using the TEBA technique fulfils its theoretical advantages over single dose intraoperative radiotherapy by decreased late toxicity and increased local tumour control.

Adolescent↗
PubMed2006

The role of high-dose therapy and autologous stem cell transplantation for pediatric bone and soft tissue sarcomas.

The prognosis for children with bone and soft tissue sarcomas has significantly improved since the advent of effective multiagent chemotherapy, aggressive surgery for local disease and more precise delivery of radiotherapy doses. However, in a small proportion of patients that present with high-risk disease, long-term outcome has not substantially increased, with disease-free survival rates still in the order of 20-30%. It is therefore clear that novel therapies are needed for children with these tumors. Based on the highly chemosensitive nature of the majority of pediatric sarcomas, several small studies have been conducted to investigate the potential role of high-dose chemotherapy followed by hematopoietic stem cell reconstitution. This review will provide an overview of the current literature concerning the use of high-dose therapy with stem cell transplantation for the three main pediatric sarcomas--Ewing sarcoma, rhabdomyosarcoma and osteosarcoma.

Bone Neoplasms↗