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Adjuvant High-Dose Thiotepa and Stem Cell Rescue Associated With Conventional Chemotherapy in Relapsed Osteosarcoma

Phase 2
Completed
Conditions
Osteosarcoma
Interventions
Registration Number
NCT00978471
Lead Sponsor
Centre Leon Berard
Brief Summary

Approximately 150 new cases of osteosarcoma are reported each year in France, of which 15 to 20% are metastatic.

Further to the initial standard care, about 45% of the patients relapse within a median duration of 20 months.

Result of the OS94 study results and of the investigation performed within the CRLCC, indicate that 25 to 30 patients (children and adults) experience an osteosarcoma relapse each year in FRANCE.

According to several studies, the 5-year overall survival rate of patients in first relapse is 23-28%,with a median post relapse survival of 10 to 17 months. Multiple relapse cases are also reported in the COSS study, with a median time to second relapse of 0.8 year.

At present, there is no reference treatment for the standard care of osteosarcoma relapse in FRANCE.

Thiotepa is known for its antitumor effect in numerous malignant tumors. In 2007, a study from our institution reported that about 35% of all osteosarcoma relapses are treated with a high-dose thiotepa while the efficacy and tolerance of this therapeutic strategy have never been assessed.

These results highlight the need to the evaluate the efficacy and tolerance of this high-dose of thiotepa within a clinical trial and its inclusion in the standard care of the osteosarcoma at relapse.

Detailed Description

Despite the absence of tumor registry, approximately 150 new cases of osteosarcoma are reported each year in France (100 cases per year in children and 50 cases in adults), of which 15 to 20% are metastatic. The standardized impact rate in the world population is estimated at 3 per million inhabitants per year.

Further to the initial standard care, about 45% of the patients relapse within a median interval of 20 months (range 3 months - 10 years).

Results of the OS94 study and of the investigation performed within the CRLCC indicate that 25 to 30 patients (children and adults) experience an osteosarcoma relapse each year in FRANCE.

Results of the five major published series indicate that the 5-year overall survival rate of patients in first relapse is between 23 and 28%, with a median post-relapse survival of 10 to 17 months. Multiple relapse cases are also reported in the COSS study, with a median time to second relapse of 0.8 year.

At present, there is no reference treatment for the standard care of osteosarcoma relapse in FRANCE.

Some recommendations have been given in the OS94 protocol, but they are generally not followed or they are implemented in a heterogeneous manner.

Thiotepa (N N' N'' triethylenethiophosphoramide), an alkylating agent of the chemical family of ethylene-imines, is known for its antitumor effect in a number of malignant tumors.

Its efficacy in osteosarcoma has been reported in the literature. A retrospective study of the SFCE (French Society for Childhood Cancer, results not yet published) in 45 patients presenting with refractory osteosarcoma or relapse has shown a radiological reaction rate of 30%.

Moreover, a preliminary investigation performed by the CLB in 2007 within the framework of the SFCE study explored all relapse cases diagnosed between the beginning of 2004 and the end of 2006. Results showed that about 35% of the patients with osteosarcoma relapses are treated with high-dose thiotepa while the efficacy and tolerance of this therapeutic strategy have never been assessed.

Altogether, these results led the SFCE osteosarcoma group to propose the evaluation of the efficacy and tolerance of this high-dose thiotepa chemotherapy within a clinical trial and to include the drug in the standard care of osteosarcoma in relapse.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
44
Inclusion Criteria
  • Age > 1 year and < 50 years
  • First osteosarcoma relapse, either local or metastatic, or second relapse after exclusive surgery NB: Whenever possible, only patients with histological evidence of relapse will be included.
  • Indication for chemotherapy confirmed by a multidisciplinary committee.
  • Surgical resection of all tumor sites must be possible, either as first-line therapy or after chemotherapy.
  • Lansky score ≥ 60%, or ECOG Performance Status ≤ 2
  • ≥ 21-day interval after first-line chemotherapy
  • Blood tests, renal and liver functions within the normal range for age with, in particular, 7 days prior to study entry, blood or serum values as follows:
  • blood: neutrophil count > 1 G/L; platelets >100 G/L
  • renal: serum creatinine ≤ 1.5 x ULN depending on age; patients with serum creatinine values > 1.5 x ULN are eligible if creatinine clearance is > 70 mL/min/1.73 m²
  • liver: total bilirubin < 2 x ULN; ASAT and ALAT ≤ 5 x ULN
  • cardiac: isotopic or echographic Left Ventricular Ejection Fraction > 50 %.
  • Signed written informed consent; for children, signed consent from the patient (depending on age) and from the parents or legal representative is mandatory
  • Documented negative serum βHCG for female patients of childbearing age
  • Affiliation with health insurance.
Exclusion Criteria
  • Patients with multiple relapses for whom surgical resection seems impossible, even after chemotherapy.
  • Patients already treated with high-dose chemotherapy regimens
  • Patients with a contra-indication to the treatment proposed
  • Patients not eligible for leukapheresis
  • Two-year follow-up impossible due to social, family, geographic or psychological reasons
  • Patient included in another protocol of clinical research
  • Pregnant or lactating women.

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
experimental arm thiotepaThiotepa4 courses of conventional chemotherapy followed by high-dose Thiotepa with peripheral stem cell rescue. Surgical resection of all tumor masses will be performed as soon as possible.
Reference armThiotepa4 courses of conventional chemotherapy. Surgical resection of all tumor masses will be performed as soon as possible.
Primary Outcome Measures
NameTimeMethod
Estimate the overall survival rate24 months
Secondary Outcome Measures
NameTimeMethod
Estimate overall survival after relapse diagnosis24 months
Estimate the survival free progression after randomization24 months
Evaluate the tolerance profile of experimental treatment (hematologic toxicity)every 3 weeks
Estimate the rate of tumor response to treatment as assessed by conventional CT-scanat inclusion, day 14-day 21 after the 2nd chemotherapy cycle, before randomization, day 14-day 21 after the 4th chemotherapy cycle, after thiotepa cure and 8 weeks after the end of treatment
Estimate histological response to treatment on surgical tumor samplesIf surgery is applicable, a few weeks after thiotepa cure (12 to 17 weeks after inclusion)
Study of biological and genomic properties and analysis of angiogenic markers correlated with relapse (optional)At inclusion,at surgery , and at the end of treatment

Trial Locations

Locations (23)

CHU Besançon- Hôpital Jean Minjoz

🇫🇷

Besancon, France

Chu - Hopital Des Enfants Bordeaux

🇫🇷

Bordeaux, France

CHU Dijon Le Bocage, Hôpital d'Enfants

🇫🇷

Dijon, France

Chu Grenoble

🇫🇷

Grenoble, France

Centre Oscar Lambret

🇫🇷

Lille, France

Hôpital des Enfants de la Timone

🇫🇷

Marseille, France

Centre Léon Bérard - Institut d'Hémato-Oncologie Pediatrique

🇫🇷

LYON Cedex 08, France

Institut Paoli Calmettes

🇫🇷

Marseille, France

Chu Nantes - Hopital Meres Et Enfants

🇫🇷

Nantes, France

Centre Antoine Lacassagne

🇫🇷

Nice, France

CHU Nice, Hôpital L'Archet 2

🇫🇷

Nice, France

Institut Curie

🇫🇷

Paris, France

Hopital D'Enfants Armand Trousseau

🇫🇷

Paris, France

CHU Poitiers, site de la Milétrie

🇫🇷

Poitiers, France

CHU de SAINT-ETIENNE, Hôpital Nord

🇫🇷

Saint Priest en Jarez, France

CHU RENNES - Hôpital Sud

🇫🇷

Rennes, France

Institut de Cancérologie de l'Ouest - René Gauducheau

🇫🇷

Saint-herblain, France

Chu La Reunion

🇫🇷

Saint-denis, France

Institut Lucien Neurwith

🇫🇷

Saint-Priest-en -Jarez, France

Hopital de Hautepierre

🇫🇷

Strasbourg, France

Institut Gustave Roussy

🇫🇷

Villejuif, France

Chu Nancy - Hopital D'Enfants

🇫🇷

Vandoeuvre Les Nancy, France

Chu Toulouse - Hopital D'Enfants

🇫🇷

Toulouse, France

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