MedPath

Comparing Two Different Myeloablation Therapies in Treating Young Patients Who Are Undergoing a Stem Cell Transplant for High-Risk Neuroblastoma

Phase 3
Completed
Conditions
Localized Resectable Neuroblastoma
Localized Unresectable Neuroblastoma
Recurrent Neuroblastoma
Regional Neuroblastoma
Stage 4 Neuroblastoma
Stage 4S Neuroblastoma
Interventions
Procedure: Autologous Hematopoietic Stem Cell Transplantation
Radiation: External Beam Radiation Therapy
Biological: Filgrastim
Other: Laboratory Biomarker Analysis
Procedure: Peripheral Blood Stem Cell Transplantation
Other: Pharmacological Study
Registration Number
NCT00567567
Lead Sponsor
Children's Oncology Group
Brief Summary

This randomized phase III trial compares two different high-dose chemotherapy regimens followed by a stem cell transplant in treating younger patients with high-risk neuroblastoma. Drugs used in chemotherapy work in different ways to stop the growth of tumor cells, either by killing the cells or by stopping them from dividing. Giving combination chemotherapy before surgery may make the tumor smaller and reduce the amount of normal tissue that needs to be removed. Giving these treatments before a peripheral blood stem cell transplant helps kill any tumor cells that are in the body and helps make room in the patient?s bone marrow for new blood-forming cells (stem cells) to grow. After treatment, stem cells are collected from the patient's blood and stored. High-dose chemotherapy and radiation therapy is then given to prepare the bone marrow for the stem cell transplant. The stem cells are then returned to the patient to replace the blood-forming cells that were destroyed by the high- chemotherapy. It is not yet known which regimen of high-dose chemotherapy is more effective for patients with high-risk neuroblastoma undergoing a peripheral blood stem cell transplant.

Detailed Description

PRIMARY OBJECTIVES:

I. To improve the 3-year event-free survival (EFS) rate of high-risk neuroblastoma patients through treatment with a tandem consolidation of thiotepa/cyclophosphamide followed by carboplatin/etoposide/melphalan (CEM) as compared to single CEM consolidation.

II. To improve the rate of end-induction complete response and very good partial response, compared to historical controls, by use of a topotecan-containing induction regimen.

III. To improve the 3-year local control rate, compared to historical controls, by increasing the local dose of radiation to the residual primary tumor for patients with less than a gross total resection.

SECONDARY OBJECTIVES:

I. To evaluate the pharmacogenetic relationship of cyclophosphamide metabolizing enzymes (CYP2B6, CYP2C9, and GSTA1 genotypes) with toxicity and response following dose-intensive cyclophosphamide and topotecan induction chemotherapy.

II. To determine if resection completeness is predictive of a) local control rate; or b) EFS rate in patients with high-risk neuroblastoma.

III. To prospectively describe the complications related to efforts at local control (surgery and radiation therapy) in patients with high-risk neuroblastoma.

IV. To describe the neurologic outcome of patients with paraspinal primary neuroblastoma tumors.

V. To determine the variability of 13-cis-retinoic-acid pharmacokinetics and relationship to pharmacogenomic parameters and determine if pharmacokinetics and/or genetic variations correlate with EFS or systemic toxicity as follows: a) To determine the variability of 13-cis-retinoic-acid pharmacokinetics and relationship to pharmacogenomic parameters. b) To determine if 13-cis-retinoic-acid pharmacokinetic levels are predictive of the EFS rate or associated with systemic toxicity following 13-cis-retinoic acid. c) To determine if pharmacogenomic variations are predictive of the EFS rate or associated with systemic toxicity following 13-cis-retinoic acid.

VI. To evaluate total topotecan pharmacokinetics and correlate with patient specific data for use in an ongoing topotecan population pharmacokinetic analysis.

VII. To evaluate the presence and function of T cells capable of recognizing neuroblastoma by assessing: a) if T cells recognizing the neuroblastoma antigen, survivin, circulate at diagnosis; b) if these T cells can be expanded using autologous antigen presenting cells (APCs); c) if these T cells will kill neuroblastoma cells as detected in functional assays; and d) if the presence and activity of anti-neuroblastoma immunity is decreased by stem cell transplantation.

VIII. To characterize the recovery of T- cell numbers after myeloablative consolidation and hematopoietic stem cell transplant (HSCT) and assess the impact of tandem myeloablative consolidation on T- cell recovery.

IX. To characterize minimal residual disease burden using reverse transcriptase-polymerase chain reaction (RT-PCR) evaluation of a panel of neuroblastoma specific transcripts in patient bone marrow and peripheral blood following induction chemotherapy and after single versus tandem myeloablative chemotherapy and to evaluate impact on EFS.

X. To evaluate the EFS and overall survival (OS) rate for patients 12-18 months with Stage 4, MYCN nonamplified tumor with unfavorable histopathology or diploid DNA content or with indeterminant histology or ploidy and patients who are greater than 547 days of age with Stage 3, MYCN nonamplified tumor AND unfavorable histopathology or indeterminant histology following treatment with single myeloablative transplant.

OUTLINE:

INDUCTION CHEMOTHERAPY:

COURSES 1 AND 2: Patients receive cyclophosphamide IV over 30 minutes and topotecan hydrochloride IV over 30 minutes on days 1-5 and filgrastim (G-CSF) subcutaneously (SC) or IV beginning on day 6 and continuing until blood counts recover. Treatment repeats every 21 days for 2 courses. Patients undergo peripheral blood stem cell (PBSC) mobilization and harvest after course 2.

COURSES 3 AND 5: Patients receive cisplatin IV over 1 hour on days 1-4, etoposide IV over 1 hour on days 1-3, and G-CSF SC or IV beginning on day 5 and continuing until blood counts recover. Treatment repeats every 21 days for 2 courses. Patients undergo surgical resection of soft tissue disease after course 5 (or after course 6 if medically necessary).

COURSES 4 AND 6: Patients receive cyclophosphamide IV over 6 hours on days 1-2, doxorubicin hydrochloride IV over 24 hours on days 1-3, vincristine IV on days 1-3, and G-CSF SC or IV beginning on day 5 and continuing until blood counts recover. Treatment repeats every 21 days for 2 courses.

Patients are then stratified by initial stage of disease and MYCN status, biologic characteristics, and response to induction chemotherapy (complete response/very good partial response vs partial response vs mixed response/no response). Patients are randomized to 1 of 2 arms. Patients 12?18 months old (i.e., 365-547 days) with stage IV, MYCN nonamplified tumor with unfavorable histopathology or diploid DNA content or with indeterminant histology or ploidy AND patients who are 547 days of age with stage III, MYCN nonamplified tumor AND unfavorable histopathology or indeterminant histology will be nonrandomly assigned to Arm A. Patients begin consolidation chemotherapy no later than 8 weeks after the start of induction course 6.

CONSOLIDATION THERAPY:

ARM A (single myeloablative consolidation): Patients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous peripheral blood stem cell transplant (PBSCT) on day 0.

ARM B (tandem myeloablative consolidation): Patients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.

RADIOTHERAPY: Patients undergo external beam radiation therapy (EBRT) to primary site of disease as well as to MIBG-avid sites seen at pre-transplantation (i.e., end-induction) evaluation between 28-42 days post-transplant. Additional radiotherapy is administered to residual tumor at primary site.

MAINTENANCE THERAPY: Patients are encouraged to enroll onto Children?s Oncology Group (COG)-ANBL0032 following assessment of tumor response after completion of the consolidation phase and radiotherapy. Beginning on day 60 post-transplantation patients receive oral isotretinoin twice daily on days 1-14. Treatment repeats every 28 days for up to 6 months in the absence of disease progression or unacceptable toxicity. Patients undergo blood and tissue sample collection periodically for the following analyses; correlation between peak serum concentration level and the existence of polymorphisms, event-free survival, and toxicity rates; pharmacogenomics for uridine diphosphate (UDP) glucuronosyltransferase 1 family, polypeptide A1 (UGT1A1), UGT2B7, CYP2C8 and CYP3A7 alleles; topotecan systemic clearance; survivin-specific cytotoxic T-lymphocytes (CTLs) detected using peptide/major histocompatibility complex (MHC) tetramers in human leukocyte antigen (HLA)-A2+ patients; interferon (IFN)-gamma production in enzyme-linked immunospot (ELISPOT) assays to APCs loaded with tumor ribonucleic acid (RNA), survivin RNA, or control RNA; response of APC-stimulated CTL response to neuroblastoma cells; rate of T cell recovery; and proportion of patients with neuroblastoma detected in bone marrow and peripheral blood using RT-PCR and immunohistochemistry (IHC).

After completion of study treatment, patients are followed up periodically for 5 years and then annually for 5 years.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
665
Inclusion Criteria
  • Diagnosis of neuroblastoma or ganglioneuroblastoma by histology or as evidenced by the presence of clumps of tumor cells in bone marrow and elevated catecholamine metabolites in urine meeting any of the following criteria:

    • Patients with newly diagnosed neuroblastoma with International Neuroblastoma Staging System (INSS) stage 4 disease are eligible with the following:

      • MYCN amplification (i.e., greater than four-fold increase in MYCN signals as compared to reference signals), regardless of age or additional biologic features
      • Age > 18 months (i.e., > 547 days) regardless of biologic features
      • Age 12-18 months (i.e., 365-547 days) with none of the following three favorable biologic features (i.e., non-amplified MYCN, favorable pathology, and deoxyribonucleic acid [DNA] index > 1)
    • Patients with newly diagnosed neuroblastoma with INSS stage 3 are eligible with the following:

      • MYCN amplification (i.e., greater than four-fold increase in MYCN signals as compared to reference signals), regardless of age or additional biologic features
      • Age > 18 months (i.e., > 547 days) with unfavorable pathology, regardless of MYCN status
    • Patients with newly diagnosed INSS stage 2a or 2b with MYCN amplification (i.e., greater than four-fold increase in MYCN signals as compared to reference signals), regardless of age or additional biologic features

    • Patients with newly diagnosed INSS stage 4s with MYCN amplification (i.e., greater than four-fold increase in MYCN signals as compared to reference signals), regardless of additional biologic features

    • Patients >= 365 days initially diagnosed with INSS stage 1, 2, or 4S and who progressed to a stage 4 without interval chemotherapy

      • Must have been enrolled on COG-ANBL00B1
  • Creatinine clearance or radioisotope glomerular filtration rate ? 70mL/min OR serum creatinine based on age/gender as follows:

    • 1 month to < 6 months: 0.4 mg/dL
    • 6 months to < 1 year: 0.5 mg/dL
    • 1 to < 2 years: 0.6 mg/dL
    • 2 to < 6 years: 0.8 mg/dL
    • 6 to < 10 years: 1 mg/dL
    • 10 to < 13 years: 1.2 mg/dL
    • 10 to < 16 years: 1.5 mg/dL (male), 1.4 mg/dL (female)
    • >= 16 years: 1.7 mg/dL (male), 1.4 mg/dL (female)
  • Total bilirubin ? 1.5 times upper limit of normal (ULN) for age

  • Aspartate aminotransferase (AST) or alanine aminotransferase (ALT) < 10 times ULN for age

  • Not pregnant or nursing

  • Negative pregnancy test

  • Shortening fraction >= 27% by echocardiogram (ECHO) OR left ventricular ejection fraction (LVEF) >= 50% by radionuclide angiogram

  • No known contraindication (e.g., size, weight or physical condition) to peripheral blood stem cell collection

  • No prior systemic therapy except for localized emergency radiation to sites of life-threatening or function-threatening disease

  • No more than one course of chemotherapy per low- or intermediate-risk neuroblastoma therapy prior to determination of MYCN amplification and histology

Exclusion Criteria

Not provided

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
Consolidation Arm A: single myeloablative consolidationAutologous Hematopoietic Stem Cell TransplantationPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationEtoposidePatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationExternal Beam Radiation TherapyPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationFilgrastimPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationLaboratory Biomarker AnalysisPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationPharmacological StudyPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationTopotecan HydrochloridePatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationVincristine Sulfate LiposomePatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationAutologous Hematopoietic Stem Cell TransplantationPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationPeripheral Blood Stem Cell TransplantationPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationLaboratory Biomarker AnalysisPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationExternal Beam Radiation TherapyPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationFilgrastimPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationIsotretinoinPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationMelphalanPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationPeripheral Blood Stem Cell TransplantationPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationPharmacological StudyPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationTopotecan HydrochloridePatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationVincristine Sulfate LiposomePatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationCarboplatinPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationCyclophosphamidePatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationCisplatinPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationDoxorubicin HydrochloridePatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationIsotretinoinPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm A: single myeloablative consolidationMelphalanPatients receive melphalan IV over 15-30 minutes on days -7 to -5, etoposide IV over 24 hours and carboplatin IV over 24 hours on days -7 to -4, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationCarboplatinPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationCisplatinPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationCyclophosphamidePatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationEtoposidePatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationDoxorubicin HydrochloridePatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Consolidation Arm B: tandem myeloablative consolidationThiotepaPatients receive thiotepa IV over 2 hours on days -7 to -5, cyclophosphamide IV over 1 hour on days -5 to -2, and G-CSF SC or IV beginning on day 0 and continuing until blood counts recover. Following clinical recovery from initial myeloablative therapy, patients also receive melphalan, etoposide, and carboplatin as in Arm A. Patients undergo autologous PBSCT on day 0.
Primary Outcome Measures
NameTimeMethod
Incidence Rate of Local RecurrenceUp to 3 years

Cumulative incidence rate of local recurrence comparison between ANBL0532 patients randomized or assigned to receive single CEM transplant and boost radiation versus the historical A3973 patients who were transplanted and received boost radiation.

Event-free Survival RateThree years, from time of randomization

Comparison of EFS curves, starting from the time of randomization, by treatment group (single CEM vs. tandem CEM)

Response After Induction TherapyStudy enrollment to the end of induction therapy

Per the International Response Criteria: measurable tumor defined as product of longest x widest perpendicular diameter. Elevated catecholamine levels, tumor cell invasion of bone marrow also considered measurable tumor. Complete Response (CR)-no evidence of primary tumor or metastases. Very Good Partial Response (VGPR)-\>90% reduction of primary tumor; no metastases; no new bone lesions, all pre-existing lesions improved. Partial Response (PR)-50-90% reduction of primary tumor; \>50% reduction in measurable sites of metastases; 0-1 bone marrow samples with tumor; number of positive bone sites decreased by \>50%. Mixed Response (MR)-\>50% reduction of any measurable lesion (primary or metastases) with \<50% reduction in other sites; no new lesions; \<25% increase in any existing lesion. No Response (NR)-no new lesions; \<50% reduction but \<25% increase in any existing legions. Progressive Disease (PD)-any new/increased measurable lesion by \>25%; previous negative marrow positive.

Secondary Outcome Measures
NameTimeMethod
Surgical ResponseUp to 3 years

Percentage of patients who achieved a surgical complete resection

Type of Surgical or Radiotherapy ComplicationUp to 3 years

The Percentage of patients who experienced surgical or radiotherapy complications will be calculated. The complications are: bowel obstruction, chylous leaf, renal injury/atrophy/loss and diarrhea.

Duration of Greater Than or Equal to Grade 3 NeutropeniaThrough completion of a participant's first cycle during induction, including treatment delays, assessed up to 39 days

A logistic regression model will be used to test the ability of the number of days of neutropenia to predict the presence of a polymorphism.

Duration of Greater Than or Equal to Grade 3 ThrombocytopeniaThrough completion of a participant's first cycle during induction, including treatment delays, assessed up to 46 days

A logistic regression model will be used to test the ability of the number of days of thrombocytopenia to predict the presence of a polymorphism.

Pharmacogenetic Variants in Patients Enrolled on Either A3973, ANBL0032, ANBL0931, ANBL0532 and Future High Risk StudiesAt baseline

To determine if pharmacogenomic variations are predictive of EFS, a logrank test comparison of patients with vs without a given polymorphism will be made. A Fisher's exact test will test for association of the presence of a polymorphism with the occurrence of systemic toxicity (CTC grade 3 or 4 skin, hypercalcemia, or hepatic toxicity). These tests will be performed for UGT1A1, UGT2B7, CYP2C8 and CYP3A7 alleles.

Proportion of Patients With a PolymorphismThrough completion of a participant's first two cycles during induction, including treatment delays, assessed up to 69 days

A chi-square test will be used to test whether the response rate after two cycles of induction therapy and the presence of a polymorphism are independent in the study population.

Intraspinal ExtensionUp to 5 years

Percentage of patients with primary tumors with intraspinal extension.

Peak Serum Concentration of Isotretinoin in Patients Enrolled on Either A3973, ANBL0032, ANBL0931, ANBL0532 and Future High Risk StudiesDay 1 of each course

Median peak serum concentration level of isotretinoin for patients enrolled on ANBL0532

Topotecan Systemic ClearanceDay 1 of courses 1-2

Median topotecan systemic clearance for courses 1 and 2.

Presence and Function of T Cells Capable of Recognizing NeuroblastomaUp to 6 months (end of therapy)
Enumeration of Peripheral Blood Cluster of Differentiation (CD)3, CD4, and CD8 CellsUp to 6 months after completion of assigned myeloablation therapy

A descriptive comparison of the median number of T-cells (CD3, CD4, CD8) between treatment arms (single vs. tandem myeloablative regimens) will be performed.

Proportion of Patients With Neuroblastoma Detected in Bone Marrow and Peripheral Blood Using RT-PCR TechniqueBaseline

Will be calculated overall and by treatment arm.

EFS Pts Non-randomly Assigned to Single CEM (12-18 Mths, Stg. 4, MYCN Nonamplified Tumor/Unfavorable or Indeterminant Histopathology/Diploid DNA Content & Pts>547 Days, Stg.3, MYCN Nonamplified Tumor AND Unfavorable or Indeterminant Histopathology).Up to 3 years

Kaplan-Meier curves of EFS will be plotted, and the proportion of responders to induction therapy will be tabulated.

OS in Patients 12-18 Months, Stage 4, MYCN Nonamplified Tumor/Unfavorable Histopathology/Diploid DNA Content/Indeterminant Histology/Ploidy and Patients > 547 Days, Stage 3, MYCN Nonamplified Tumor AND Unfavorable Histopathology/Indeterminant HistologyUp to 3 years

Kaplan-Meier curves of OS will be plotted, and the proportion of responders to induction therapy will be tabulated.

Trial Locations

Locations (190)

Cedars Sinai Medical Center

🇺🇸

Los Angeles, California, United States

Valley Children's Hospital

🇺🇸

Madera, California, United States

Children's Healthcare of Atlanta - Egleston

🇺🇸

Atlanta, Georgia, United States

Eastern Maine Medical Center

🇺🇸

Bangor, Maine, United States

University of Illinois

🇺🇸

Chicago, Illinois, United States

Rainbow Babies and Childrens Hospital

🇺🇸

Cleveland, Ohio, United States

University of Miami Miller School of Medicine-Sylvester Cancer Center

🇺🇸

Miami, Florida, United States

Nicklaus Children's Hospital

🇺🇸

Miami, Florida, United States

Saint Vincent Hospital and Health Care Center

🇺🇸

Indianapolis, Indiana, United States

Nevada Cancer Research Foundation CCOP

🇺🇸

Las Vegas, Nevada, United States

IWK Health Centre

🇨🇦

Halifax, Nova Scotia, Canada

Saint Mary's Hospital

🇺🇸

West Palm Beach, Florida, United States

Indiana University/Melvin and Bren Simon Cancer Center

🇺🇸

Indianapolis, Indiana, United States

University of Chicago Comprehensive Cancer Center

🇺🇸

Chicago, Illinois, United States

Saint Jude Midwest Affiliate

🇺🇸

Peoria, Illinois, United States

Maine Children's Cancer Program

🇺🇸

Scarborough, Maine, United States

Emory University Hospital/Winship Cancer Institute

🇺🇸

Atlanta, Georgia, United States

Blank Children's Hospital

🇺🇸

Des Moines, Iowa, United States

Johns Hopkins All Children's Hospital

🇺🇸

Saint Petersburg, Florida, United States

Riley Hospital for Children

🇺🇸

Indianapolis, Indiana, United States

Southern Illinois University School of Medicine

🇺🇸

Springfield, Illinois, United States

Mercy Children's Hospital

🇺🇸

Toledo, Ohio, United States

Hospital for Sick Children

🇨🇦

Toronto, Ontario, Canada

Victoria Hospital

🇨🇦

London, Ontario, Canada

McMaster Children's Hospital at Hamilton Health Sciences

🇨🇦

Hamilton, Ontario, Canada

San Jorge Children's Hospital

🇵🇷

San Juan, Puerto Rico

Kingston Health Sciences Centre

🇨🇦

Kingston, Ontario, Canada

Children's Hospital of Eastern Ontario

🇨🇦

Ottawa, Ontario, Canada

Starship Children's Hospital

🇳🇿

Grafton, Auckland, New Zealand

UCSF Medical Center-Mount Zion

🇺🇸

San Francisco, California, United States

UCSF Medical Center-Parnassus

🇺🇸

San Francisco, California, United States

University of Minnesota/Masonic Cancer Center

🇺🇸

Minneapolis, Minnesota, United States

Duke University Medical Center

🇺🇸

Durham, North Carolina, United States

Vanderbilt University/Ingram Cancer Center

🇺🇸

Nashville, Tennessee, United States

Methodist Children's Hospital of South Texas

🇺🇸

San Antonio, Texas, United States

New York Medical College

🇺🇸

Valhalla, New York, United States

Princess Margaret Hospital for Children

🇦🇺

Perth, Western Australia, Australia

University of Rochester

🇺🇸

Rochester, New York, United States

Phoenix Childrens Hospital

🇺🇸

Phoenix, Arizona, United States

Wayne State University/Karmanos Cancer Institute

🇺🇸

Detroit, Michigan, United States

University of Oklahoma Health Sciences Center

🇺🇸

Oklahoma City, Oklahoma, United States

Ascension Saint John Hospital

🇺🇸

Detroit, Michigan, United States

Children's Hospital of Alabama

🇺🇸

Birmingham, Alabama, United States

University of Alabama at Birmingham Cancer Center

🇺🇸

Birmingham, Alabama, United States

Rocky Mountain Hospital for Children-Presbyterian Saint Luke's Medical Center

🇺🇸

Denver, Colorado, United States

Mayo Clinic

🇺🇸

Rochester, Minnesota, United States

Legacy Emanuel Hospital and Health Center

🇺🇸

Portland, Oregon, United States

Oregon Health and Science University

🇺🇸

Portland, Oregon, United States

Royal Children's Hospital-Brisbane

🇦🇺

Herston, Queensland, Australia

Saint Joseph's Hospital/Children's Hospital-Tampa

🇺🇸

Tampa, Florida, United States

Children's Hospital Colorado

🇺🇸

Aurora, Colorado, United States

Children's Hospital of Wisconsin

🇺🇸

Milwaukee, Wisconsin, United States

Kaiser Permanente Downey Medical Center

🇺🇸

Downey, California, United States

City of Hope Comprehensive Cancer Center

🇺🇸

Duarte, California, United States

Kaiser Permanente-Oakland

🇺🇸

Oakland, California, United States

Lucile Packard Children's Hospital Stanford University

🇺🇸

Palo Alto, California, United States

Children's National Medical Center

🇺🇸

Washington, District of Columbia, United States

Golisano Children's Hospital of Southwest Florida

🇺🇸

Fort Myers, Florida, United States

Sacred Heart Hospital

🇺🇸

Pensacola, Florida, United States

University of Maryland/Greenebaum Cancer Center

🇺🇸

Baltimore, Maryland, United States

Johns Hopkins University/Sidney Kimmel Cancer Center

🇺🇸

Baltimore, Maryland, United States

Sinai Hospital of Baltimore

🇺🇸

Baltimore, Maryland, United States

Walter Reed National Military Medical Center

🇺🇸

Bethesda, Maryland, United States

C S Mott Children's Hospital

🇺🇸

Ann Arbor, Michigan, United States

Hurley Medical Center

🇺🇸

Flint, Michigan, United States

Children's Hospitals and Clinics of Minnesota - Minneapolis

🇺🇸

Minneapolis, Minnesota, United States

Washington University School of Medicine

🇺🇸

Saint Louis, Missouri, United States

Dartmouth Hitchcock Medical Center

🇺🇸

Lebanon, New Hampshire, United States

Newark Beth Israel Medical Center

🇺🇸

Newark, New Jersey, United States

Laura and Isaac Perlmutter Cancer Center at NYU Langone

🇺🇸

New York, New York, United States

Mount Sinai Hospital

🇺🇸

New York, New York, United States

NYP/Columbia University Medical Center/Herbert Irving Comprehensive Cancer Center

🇺🇸

New York, New York, United States

Sanford Broadway Medical Center

🇺🇸

Fargo, North Dakota, United States

The Toledo Hospital/Toledo Children's Hospital

🇺🇸

Toledo, Ohio, United States

Geisinger Medical Center

🇺🇸

Danville, Pennsylvania, United States

Lehigh Valley Hospital - Muhlenberg

🇺🇸

Bethlehem, Pennsylvania, United States

Saint Christopher's Hospital for Children

🇺🇸

Philadelphia, Pennsylvania, United States

East Tennessee Childrens Hospital

🇺🇸

Knoxville, Tennessee, United States

Carilion Clinic Children's Hospital

🇺🇸

Roanoke, Virginia, United States

Seattle Children's Hospital

🇺🇸

Seattle, Washington, United States

British Columbia Children's Hospital

🇨🇦

Vancouver, British Columbia, Canada

Centre Hospitalier Universitaire Sainte-Justine

🇨🇦

Montreal, Quebec, Canada

Centre Hospitalier Universitaire de Quebec

🇨🇦

Quebec, Canada

Swiss Pediatric Oncology Group - Bern

🇨🇭

Bern, Switzerland

University of Arkansas for Medical Sciences

🇺🇸

Little Rock, Arkansas, United States

Children's Hospital and Research Center at Oakland

🇺🇸

Oakland, California, United States

USA Health Strada Patient Care Center

🇺🇸

Mobile, Alabama, United States

Children's Hospital Los Angeles

🇺🇸

Los Angeles, California, United States

The University of Arizona Medical Center-University Campus

🇺🇸

Tucson, Arizona, United States

Nemours Children's Clinic-Jacksonville

🇺🇸

Jacksonville, Florida, United States

Loma Linda University Medical Center

🇺🇸

Loma Linda, California, United States

Miller Children's and Women's Hospital Long Beach

🇺🇸

Long Beach, California, United States

Children's Hospital of Orange County

🇺🇸

Orange, California, United States

Rady Children's Hospital - San Diego

🇺🇸

San Diego, California, United States

Harbor-University of California at Los Angeles Medical Center

🇺🇸

Torrance, California, United States

Memorial Regional Hospital/Joe DiMaggio Children's Hospital

🇺🇸

Hollywood, Florida, United States

University of Connecticut

🇺🇸

Farmington, Connecticut, United States

Connecticut Children's Medical Center

🇺🇸

Hartford, Connecticut, United States

Alfred I duPont Hospital for Children

🇺🇸

Wilmington, Delaware, United States

Broward Health Medical Center

🇺🇸

Fort Lauderdale, Florida, United States

Lee Memorial Health System

🇺🇸

Fort Myers, Florida, United States

Augusta University Medical Center

🇺🇸

Augusta, Georgia, United States

Nemours Children's Clinic - Pensacola

🇺🇸

Pensacola, Florida, United States

Memorial Health University Medical Center

🇺🇸

Savannah, Georgia, United States

Lurie Children's Hospital-Chicago

🇺🇸

Chicago, Illinois, United States

Loyola University Medical Center

🇺🇸

Maywood, Illinois, United States

University of Iowa/Holden Comprehensive Cancer Center

🇺🇸

Iowa City, Iowa, United States

Floating Hospital for Children at Tufts Medical Center

🇺🇸

Boston, Massachusetts, United States

University of Kansas Cancer Center

🇺🇸

Kansas City, Kansas, United States

Massachusetts General Hospital Cancer Center

🇺🇸

Boston, Massachusetts, United States

University of Kentucky/Markey Cancer Center

🇺🇸

Lexington, Kentucky, United States

Dana-Farber Cancer Institute

🇺🇸

Boston, Massachusetts, United States

Kalamazoo Center for Medical Studies

🇺🇸

Kalamazoo, Michigan, United States

Spectrum Health at Butterworth Campus

🇺🇸

Grand Rapids, Michigan, United States

University of Massachusetts Medical School

🇺🇸

Worcester, Massachusetts, United States

University of Missouri - Ellis Fischel

🇺🇸

Columbia, Missouri, United States

Mercy Hospital Saint Louis

🇺🇸

Saint Louis, Missouri, United States

University of Mississippi Medical Center

🇺🇸

Jackson, Mississippi, United States

Helen DeVos Children's Hospital at Spectrum Health

🇺🇸

Grand Rapids, Michigan, United States

Michigan State University Clinical Center

🇺🇸

East Lansing, Michigan, United States

Brooklyn Hospital Center

🇺🇸

Brooklyn, New York, United States

Bronson Methodist Hospital

🇺🇸

Kalamazoo, Michigan, United States

Children's Hospital and Medical Center of Omaha

🇺🇸

Omaha, Nebraska, United States

University of Nebraska Medical Center

🇺🇸

Omaha, Nebraska, United States

Saint Peter's University Hospital

🇺🇸

New Brunswick, New Jersey, United States

Hackensack University Medical Center

🇺🇸

Hackensack, New Jersey, United States

Morristown Medical Center

🇺🇸

Morristown, New Jersey, United States

Rutgers Cancer Institute of New Jersey-Robert Wood Johnson University Hospital

🇺🇸

New Brunswick, New Jersey, United States

University of New Mexico Cancer Center

🇺🇸

Albuquerque, New Mexico, United States

Saint Joseph's Regional Medical Center

🇺🇸

Paterson, New Jersey, United States

State University of New York Upstate Medical University

🇺🇸

Syracuse, New York, United States

Albany Medical Center

🇺🇸

Albany, New York, United States

Overlook Hospital

🇺🇸

Summit, New Jersey, United States

Penn State Children's Hospital

🇺🇸

Hershey, Pennsylvania, United States

Mission Hospital Inc-Memorial Campus

🇺🇸

Asheville, North Carolina, United States

Roswell Park Cancer Institute

🇺🇸

Buffalo, New York, United States

University of Toledo

🇺🇸

Toledo, Ohio, United States

The Steven and Alexandra Cohen Children's Medical Center of New York

🇺🇸

New Hyde Park, New York, United States

Carolinas Medical Center/Levine Cancer Institute

🇺🇸

Charlotte, North Carolina, United States

Cleveland Clinic Foundation

🇺🇸

Cleveland, Ohio, United States

Cincinnati Children's Hospital Medical Center

🇺🇸

Cincinnati, Ohio, United States

Dayton Children's Hospital

🇺🇸

Dayton, Ohio, United States

Nationwide Children's Hospital

🇺🇸

Columbus, Ohio, United States

Legacy Emanuel Children's Hospital

🇺🇸

Portland, Oregon, United States

Children's Hospital of Philadelphia

🇺🇸

Philadelphia, Pennsylvania, United States

Children's Hospital of Pittsburgh of UPMC

🇺🇸

Pittsburgh, Pennsylvania, United States

Prisma Health Richland Hospital

🇺🇸

Columbia, South Carolina, United States

Providence Sacred Heart Medical Center and Children's Hospital

🇺🇸

Spokane, Washington, United States

Sanford USD Medical Center - Sioux Falls

🇺🇸

Sioux Falls, South Dakota, United States

BI-LO Charities Children's Cancer Center

🇺🇸

Greenville, South Carolina, United States

Greenville Cancer Treatment Center

🇺🇸

Greenville, South Carolina, United States

T C Thompson Children's Hospital

🇺🇸

Chattanooga, Tennessee, United States

Driscoll Children's Hospital

🇺🇸

Corpus Christi, Texas, United States

UT Southwestern/Simmons Cancer Center-Dallas

🇺🇸

Dallas, Texas, United States

Cook Children's Medical Center

🇺🇸

Fort Worth, Texas, United States

Primary Children's Hospital

🇺🇸

Salt Lake City, Utah, United States

University of Virginia Cancer Center

🇺🇸

Charlottesville, Virginia, United States

Covenant Children's Hospital

🇺🇸

Lubbock, Texas, United States

University of Vermont and State Agricultural College

🇺🇸

Burlington, Vermont, United States

University of Texas Health Science Center at San Antonio

🇺🇸

San Antonio, Texas, United States

Inova Fairfax Hospital

🇺🇸

Falls Church, Virginia, United States

Naval Medical Center - Portsmouth

🇺🇸

Portsmouth, Virginia, United States

Madigan Army Medical Center

🇺🇸

Tacoma, Washington, United States

West Virginia University Charleston Division

🇺🇸

Charleston, West Virginia, United States

Marshfield Medical Center-Marshfield

🇺🇸

Marshfield, Wisconsin, United States

Westmead Hospital

🇦🇺

Westmead, New South Wales, Australia

Saint Vincent Hospital Cancer Center Green Bay

🇺🇸

Green Bay, Wisconsin, United States

Royal Brisbane and Women's Hospital

🇦🇺

Herston, Queensland, Australia

University of Wisconsin Hospital and Clinics

🇺🇸

Madison, Wisconsin, United States

The Children's Hospital at Westmead

🇦🇺

Westmead, New South Wales, Australia

CancerCare Manitoba

🇨🇦

Winnipeg, Manitoba, Canada

Alberta Children's Hospital

🇨🇦

Calgary, Alberta, Canada

University of Alberta Hospital

🇨🇦

Edmonton, Alberta, Canada

University of California Davis Comprehensive Cancer Center

🇺🇸

Sacramento, California, United States

Florida Hospital Orlando

🇺🇸

Orlando, Florida, United States

Nemours Children's Clinic - Orlando

🇺🇸

Orlando, Florida, United States

Norton Children's Hospital

🇺🇸

Louisville, Kentucky, United States

UF Cancer Center at Orlando Health

🇺🇸

Orlando, Florida, United States

Wake Forest University Health Sciences

🇺🇸

Winston-Salem, North Carolina, United States

UNC Lineberger Comprehensive Cancer Center

🇺🇸

Chapel Hill, North Carolina, United States

Children's Mercy Hospitals and Clinics

🇺🇸

Kansas City, Missouri, United States

Medical University of South Carolina

🇺🇸

Charleston, South Carolina, United States

Tulane University Health Sciences Center

🇺🇸

New Orleans, Louisiana, United States

University of Florida Health Science Center - Gainesville

🇺🇸

Gainesville, Florida, United States

Children's Hospital New Orleans

🇺🇸

New Orleans, Louisiana, United States

Dell Children's Medical Center of Central Texas

🇺🇸

Austin, Texas, United States

Virginia Commonwealth University/Massey Cancer Center

🇺🇸

Richmond, Virginia, United States

Arkansas Children's Hospital

🇺🇸

Little Rock, Arkansas, United States

Novant Health Presbyterian Medical Center

🇺🇸

Charlotte, North Carolina, United States

Children's Hospital

🇨🇦

London, Ontario, Canada

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