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Inhaled Sargramostim in Treating Patients With First Pulmonary (Lung) Recurrence of Osteosarcoma

Phase 2
Completed
Conditions
Sarcoma
Metastatic Cancer
Interventions
Biological: sargramostim
Procedure: conventional surgery
Registration Number
NCT00066365
Lead Sponsor
Children's Oncology Group
Brief Summary

RATIONALE: Inhaling aerosolized sargramostim before and after surgery may interfere with the growth of tumor cells and shrink the tumor so that it can be removed during surgery. Sargramostim may then kill any tumor cells remaining after surgery. This may be an effective treatment for osteosarcoma that has spread to the lung.

PURPOSE: This phase II trial is studying how well inhaled sargramostim works in treating patients who are undergoing surgery for the first recurrence of osteosarcoma that has spread to the lung.

Detailed Description

OBJECTIVES:

Primary

* Assess the histological findings from patients with first pulmonary recurrence of osteosarcoma who undergo resection of pulmonary metastases after treatment with 2 courses of aerosolized sargramostim (GM-CSF).

* Determine the event-free survival of patients treated with this drug.

* Determine whether the maximum tolerated dose in the trial of inhaled GM-CSF in adult patients with melanoma is tolerable in pediatric patients.

Secondary

* Determine the effect of specific thoracic surgical management on outcome in patients treated with this drug.

OUTLINE: This is a multicenter, dose escalation study. Patients are assigned to 1 of 2 groups according to the extent of pulmonary recurrence (unilateral or bilateral).

* Group I (unilateral recurrence):

* Initial inhalation therapy: Patients receive inhaled sargramostim (GM-CSF) twice daily on days 1-7. Treatment repeats every other week every 14 days for a total of 2 courses.

* Thoracotomy: Patients undergo thoracotomy on day 22.

* Post-thoracotomy inhalation therapy: Beginning on day 29, or as soon as possible thereafter, patients resume inhalation therapy as above for up to 12 additional courses.

* Group II (bilateral recurrence): Patients may be enrolled on study either before or after the first thoracotomy.

* First thoracotomy: Patients undergo unilateral thoracotomy.

* Initial inhalation therapy: Patients receive inhaled GM-CSF, as soon as possible after recovery from first thoracotomy, twice daily on days 1-7. Treatment repeats every other week every 14 days for a total of 2 courses.

* Contralateral thoracotomy: Patients undergo contralateral thoracotomy on day 22.

* Post-thoracotomy inhalation therapy: Beginning on day 29, or as soon as possible, patients resume inhalation therapy as above for up to 12 additional courses.

Treatment in both groups continues in the absence of disease progression or unacceptable toxicity.

Patients are followed every 2 months for 1 year, every 4 months for 1 year, every 6 months for 3 years, and then annually thereafter.

PROJECTED ACCRUAL: A total of 40 patients will be accrued for this study within 1.6-2 years.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
49
Inclusion Criteria

Not provided

Exclusion Criteria

Not provided

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
Group 1 (unilateral recurrence) - Sargramostim and thoractomyconventional surgeryPatients receive initial inhalation therapy inhaled sargramostim (GM-CSF) twice daily on days 1-7. Treatment repeats every other week every 14 days for a total of 2 courses. Patients undergo surgical procedure thoracotomy on day 22. Beginning on day 29, or as soon as possible thereafter, patients begin post-thoracotomy inhalation therapy for up to 12 additional courses. Treatment continues in the absence of disease progression or unacceptable toxicity. Patients are followed every 2 months for 1 year, every 4 months for 1 year, every 6 months for 3 years, and then annually thereafter.
Group 1 (unilateral recurrence) - Sargramostim and thoractomysargramostimPatients receive initial inhalation therapy inhaled sargramostim (GM-CSF) twice daily on days 1-7. Treatment repeats every other week every 14 days for a total of 2 courses. Patients undergo surgical procedure thoracotomy on day 22. Beginning on day 29, or as soon as possible thereafter, patients begin post-thoracotomy inhalation therapy for up to 12 additional courses. Treatment continues in the absence of disease progression or unacceptable toxicity. Patients are followed every 2 months for 1 year, every 4 months for 1 year, every 6 months for 3 years, and then annually thereafter.
Group 2 (bilateral recurrence) - Sargramostim and thoractomyconventional surgeryPatients may be enrolled on study either before or after the first thoracotomy procedure. For the first thoracotomy, patients undergo surgical procedure unilateral thoracotomy. Patients receive initial inhalation therapy inhaled GM-CSF, as soon as possible after recovery from first thoracotomy, twice daily on days 1-7. Treatment repeats every other week every 14 days for a total of 2 courses. Patients undergo surgical procedure contralateral thoracotomy on day 22. Beginning on day 29, or as soon as possible, patients begin post-thoracotomy inhalation therapy as above for up to 12 additional courses. Treatment continues in the absence of disease progression or unacceptable toxicity. Patients are followed every 2 months for 1 year, every 4 months for 1 year, every 6 months for 3 years, and then annually thereafter.
Group 2 (bilateral recurrence) - Sargramostim and thoractomysargramostimPatients may be enrolled on study either before or after the first thoracotomy procedure. For the first thoracotomy, patients undergo surgical procedure unilateral thoracotomy. Patients receive initial inhalation therapy inhaled GM-CSF, as soon as possible after recovery from first thoracotomy, twice daily on days 1-7. Treatment repeats every other week every 14 days for a total of 2 courses. Patients undergo surgical procedure contralateral thoracotomy on day 22. Beginning on day 29, or as soon as possible, patients begin post-thoracotomy inhalation therapy as above for up to 12 additional courses. Treatment continues in the absence of disease progression or unacceptable toxicity. Patients are followed every 2 months for 1 year, every 4 months for 1 year, every 6 months for 3 years, and then annually thereafter.
Primary Outcome Measures
NameTimeMethod
Status of FAS Ligand in Pre-chemotherapy Sample29 days after start of protocol therapy

FAS ligand (FASL) is a homotrimeric type II transmembrane protein expressed on cytotoxic T lymphocytes. The Cluster of Differentiation 1a (CD1a) status is measured in Immunohistochemistry (IHC) categories.

Presence of FAS in Pre-chemotherapy Sample29 days after start of protocol therapy

FAS/APO-1 is a transmembrane receptor. The presence is measured in Immunohistochemistry (IHC) categories.

FAS Ligand in Post Chemotherapy Sample29 days after start of protocol therapy

FAS ligand or FASL is a homotrimeric type II transmembrane protein expressed on cytotoxic T lymphocytes. The presence is measured in Immunohistochemistry (IHC) categories.

FAS Status in Post Chemotherapy Sample29 days after start of protocol therapy

FAS/APO-1 is a transmembrane receptor. The presence is measured in Immunohistochemistry (IHC) categories.

CD1a Status in Pre Chemotherapy Sample29 days after start of protocol therapy

CD1a (Cluster of Differentiation 1a) is a human protein encoded by the CD1A gene, presence is measured by positivity.

CD1a Status in Post Chemotherapy Sample29 days after start of protocol therapy

CD1a (Cluster of Differentiation 1a) is a human protein encoded by the CD1A gene, presence is measured by positivity.

S100 Status in Pre Chemotherapy Sample29 days after start of protocol therapy

The S-100 proteins are a family of low-molecular-weight proteins characterized by two calcium-binding sites that have helix-loop-helix ("EF-hand type") conformation.

S100 Status in Post Chemotherapy Sample29 days after start of protocol therapy

The S-100 proteins are a family of low-molecular-weight proteins characterized by two calcium-binding sites that have helix-loop-helix ("EF-hand type") conformation.

Clusterin Status in Pre Chemotherapy Sample29 days after start of protocol therapy

The protein encoded by this gene can under some stress conditions also be found in the cell cytosol. It has been suggested to be involved in several basic biological events such as cell death, tumor progression, and neurodegenerative disorders.

Clusterin Status in Post Chemotherapy Sample29 days after start of protocol therapy
Event Free Survival (EFS)Time of enrollment to Event or 5 years from enrollment, whichever occurs first

EFS defined as the time from enrollment on the study until disease progression, occurrence of a second malignant neoplasm (SMN), death or last contact, whichever comes first. Disease progression, occurrence of a SMN or death will be considered an analytic even. In all other cases, the patient will be considered censored at last contact.

Feasibility SuccessEnrollment through 21 days of protocol therapy

Feasibility success defined as received 21 days of protocol therapy, did not experience grade III or grade IV toxicity according to Common Toxicity Criteria for Adverse Events (CTCAE) version 3 and rendered surgically free of disease in the lungs.

Secondary Outcome Measures
NameTimeMethod

Trial Locations

Locations (93)

Sidney Kimmel Comprehensive Cancer Center at Johns Hopkins

🇺🇸

Baltimore, Maryland, United States

Miami Children's Hospital

🇺🇸

Miami, Florida, United States

Dana-Farber/Harvard Cancer Center at Dana Farber Cancer Institute

🇺🇸

Boston, Massachusetts, United States

Children's Hospital and Regional Medical Center - Seattle

🇺🇸

Seattle, Washington, United States

Arkansas Cancer Research Center at University of Arkansas for Medical Sciences

🇺🇸

Little Rock, Arkansas, United States

Arizona Cancer Center at University of Arizona Health Sciences Center

🇺🇸

Tucson, Arizona, United States

Lee Cancer Care of Lee Memorial Health System

🇺🇸

Fort Myers, Florida, United States

Lombardi Comprehensive Cancer Center at Georgetown University Medical Center

🇺🇸

Washington, District of Columbia, United States

Kaplan Cancer Center at St. Mary's Medical Center

🇺🇸

West Palm Beach, Florida, United States

All Children's Hospital

🇺🇸

St. Petersburg, Florida, United States

Herbert Irving Comprehensive Cancer Center at Columbia University Medical Center

🇺🇸

New York, New York, United States

Palmetto Health South Carolina Cancer Center

🇺🇸

Columbia, South Carolina, United States

Children's Hospital of Pittsburgh

🇺🇸

Pittsburgh, Pennsylvania, United States

CCOP - Scott and White Hospital

🇺🇸

Temple, Texas, United States

Simmons Comprehensive Cancer Center at University of Texas Southwestern Medical Center - Dallas

🇺🇸

Dallas, Texas, United States

East Tennessee Children's Hospital

🇺🇸

Knoxville, Tennessee, United States

Cook Children's Medical Center - Fort Worth

🇺🇸

Fort Worth, Texas, United States

St. Vincent Hospital Regional Cancer Center

🇺🇸

Green Bay, Wisconsin, United States

Fletcher Allen Health Care - University Health Center Campus

🇺🇸

Burlington, Vermont, United States

University of Texas Health Science Center at San Antonio

🇺🇸

San Antonio, Texas, United States

Lurleen Wallace Comprehensive Cancer at University of Alabama - Birmingham

🇺🇸

Birmingham, Alabama, United States

Phoenix Children's Hospital

🇺🇸

Phoenix, Arizona, United States

Barbara Ann Karmanos Cancer Institute

🇺🇸

Detroit, Michigan, United States

Hopital Sainte Justine

🇨🇦

Montreal, Quebec, Canada

Southern California Permanente Medical Group

🇺🇸

Downey, California, United States

University of Miami Sylvester Comprehensive Cancer Center - Miami

🇺🇸

Miami, Florida, United States

Children's Hospital and Research Center Oakland

🇺🇸

Oakland, California, United States

Curtis and Elizabeth Anderson Cancer Institute at Memorial Health University Medical Center

🇺🇸

Savannah, Georgia, United States

Jonathan Jaques Children's Cancer Center at Miller Children's Hospital

🇺🇸

Long Beach, California, United States

Baptist-South Miami Regional Cancer Program

🇺🇸

Miami, Florida, United States

Stanford Cancer Center

🇺🇸

Stanford, California, United States

Loma Linda University Cancer Institute at Loma Linda University Medical Center

🇺🇸

Loma Linda, California, United States

Alfred I. duPont Hospital for Children

🇺🇸

Wilmington, Delaware, United States

Sacred Heart Cancer Center at Sacred Heart Hospital

🇺🇸

Pensacola, Florida, United States

Children's Memorial Hospital - Chicago

🇺🇸

Chicago, Illinois, United States

Indiana University Melvin and Bren Simon Cancer Center

🇺🇸

Indianapolis, Indiana, United States

Winship Cancer Institute of Emory University

🇺🇸

Atlanta, Georgia, United States

Alvin and Lois Lapidus Cancer Institute at Sinai Hospital

🇺🇸

Baltimore, Maryland, United States

Nationwide Children's Hospital

🇺🇸

Columbus, Ohio, United States

Mountain States Tumor Institute at St. Luke's Regional Medical Center

🇺🇸

Boise, Idaho, United States

Newark Beth Israel Medical Center

🇺🇸

Newark, New Jersey, United States

Akron Children's Hospital

🇺🇸

Akron, Ohio, United States

Children's Medical Center - Dayton

🇺🇸

Dayton, Ohio, United States

Butterworth Hospital at Spectrum Health

🇺🇸

Grand Rapids, Michigan, United States

Covenant Children's Hospital

🇺🇸

Lubbock, Texas, United States

University of Mississippi Cancer Clinic

🇺🇸

Jackson, Mississippi, United States

Simmons Cooper Cancer Institute

🇺🇸

Springfield, Illinois, United States

Oklahoma University Cancer Institute

🇺🇸

Oklahoma City, Oklahoma, United States

Roswell Park Cancer Institute

🇺🇸

Buffalo, New York, United States

Hackensack University Medical Center Cancer Center

🇺🇸

Hackensack, New Jersey, United States

Cancer Institute of New Jersey at UMDNJ - Robert Wood Johnson Medical School

🇺🇸

New Brunswick, New Jersey, United States

Blumenthal Cancer Center at Carolinas Medical Center

🇺🇸

Charlotte, North Carolina, United States

Cleveland Clinic Taussig Cancer Center

🇺🇸

Cleveland, Ohio, United States

Duke Comprehensive Cancer Center

🇺🇸

Durham, North Carolina, United States

Penn State Cancer Institute at Milton S. Hershey Medical Center

🇺🇸

Hershey, Pennsylvania, United States

St. Christopher's Hospital for Children

🇺🇸

Philadelphia, Pennsylvania, United States

M. D. Anderson Cancer Center at University of Texas

🇺🇸

Houston, Texas, United States

Greenville Hospital Cancer Center

🇺🇸

Greenville, South Carolina, United States

Marshfield Clinic - Marshfield Center

🇺🇸

Marshfield, Wisconsin, United States

Providence Cancer Center at Sacred Heart Medical Center

🇺🇸

Spokane, Washington, United States

Centre Hospitalier Universitaire de Quebec

🇨🇦

Quebec, Canada

McMaster Children's Hospital at Hamilton Health Sciences

🇨🇦

Hamilton, Ontario, Canada

Montreal Children's Hospital at McGill University Health Center

🇨🇦

Montreal, Quebec, Canada

CancerCare Manitoba

🇨🇦

Winnipeg, Manitoba, Canada

IWK Health Centre

🇨🇦

Halifax, Nova Scotia, Canada

San Jorge Children's Hospital

🇵🇷

Santurce, Puerto Rico

SUNY Upstate Medical University Hospital

🇺🇸

Syracuse, New York, United States

James P. Wilmot Cancer Center at University of Rochester Medical Center

🇺🇸

Rochester, New York, United States

Princess Margaret Hospital for Children

🇦🇺

Perth, Western Australia, Australia

Kansas Masonic Cancer Research Institute at the University of Kansas Medical Center

🇺🇸

Kansas City, Kansas, United States

Westmead Institute for Cancer Research at Westmead Hospital

🇦🇺

Westmead, New South Wales, Australia

St. Joseph's Cancer Institute at St. Joseph's Hospital

🇺🇸

Tampa, Florida, United States

Mayo Clinic Cancer Center

🇺🇸

Rochester, Minnesota, United States

Legacy Emanuel Hospital and Health Center and Children's Hospital

🇺🇸

Portland, Oregon, United States

Oregon Health and Science University Cancer Institute

🇺🇸

Portland, Oregon, United States

Midwest Children's Cancer Center at Children's Hospital of Wisconsin

🇺🇸

Milwaukee, Wisconsin, United States

UCSF Helen Diller Family Comprehensive Cancer Center

🇺🇸

San Francisco, California, United States

Hurley Medical Center

🇺🇸

Flint, Michigan, United States

Van Elslander Cancer Center at St. John Hospital and Medical Center

🇺🇸

Grosse Pointe Woods, Michigan, United States

Breslin Cancer Center at Ingham Regional Medical Center

🇺🇸

Lansing, Michigan, United States

Lehigh Valley Hospital - Muhlenberg

🇺🇸

Bethlehem, Pennsylvania, United States

Children's Hospital of Philadelphia

🇺🇸

Philadelphia, Pennsylvania, United States

University of California Davis Cancer Center

🇺🇸

Sacramento, California, United States

Nemours Children's Clinic

🇺🇸

Jacksonville, Florida, United States

University of Florida Shands Cancer Center

🇺🇸

Gainesville, Florida, United States

Nemours Children's Clinic - Orlando

🇺🇸

Orlando, Florida, United States

Lucille P. Markey Cancer Center at University of Kentucky

🇺🇸

Lexington, Kentucky, United States

Kosair Children's Hospital

🇺🇸

Louisville, Kentucky, United States

Children's Mercy Hospital

🇺🇸

Kansas City, Missouri, United States

University of New Mexico Cancer Center

🇺🇸

Albuquerque, New Mexico, United States

University of Wisconsin Paul P. Carbone Comprehensive Cancer Center

🇺🇸

Madison, Wisconsin, United States

Saskatoon Cancer Centre at the University of Saskatchewan

🇨🇦

Saskatoon, Saskatchewan, Canada

Children's National Medical Center

🇺🇸

Washington, District of Columbia, United States

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