NCT01193283

Brief Summary

Background:

  • Severe aplastic anemia (SAA) can lead to problems with bone marrow health and result in low blood cell counts, which require frequent transfusions. Standard treatment for SAA involves injections of antithymocyte globulin (ATG) plus cyclosporine (CsA). This regimen has been shown to improve the blood counts in about two-thirds of patients. However, the ATG/CsA regimen has the following limitations: (a) the disease can come back (relapse) in about one-third of patients who improve initially; and (b) in about 10% to 15% of cases, certain types of bone marrow cancer (such as myelodysplasia and leukemia) can develop (called evolution). Experience with other drugs in SAA such as cyclophosphamide suggests that similar response rates to ATG/CsA can be achieved with a lower risk of relapse and clonal evolution. However, cyclophosphamide was found to have significant side effects in SAA when investigated over 10 years ago due to increase risk of fungal infections.
  • Better antibiotic drugs against fungus have been developed and are widely used to treat patients who have low white blood cell counts and are at risk of developing infections. In SAA patients in particular, these newer antibiotics have had a large impact in preventing and treating fungus infections. Researchers are revisiting the use of cyclophosphamide in SAA treatment, and plan to give a lower dose of CsA in combination with the immune-suppressing drug cyclophosphamide, as well as antibiotics to protect against infections, as a possible treatment for the disease. Objectives: \- To determine the safety and effectiveness of the combination of cyclophosphamide and cyclosporine in treating severe aplastic anemia that has not been treated with immunosuppressive therapy.

Trial Health

87
On Track

Trial Health Score

Automated assessment based on enrollment pace, timeline, and geographic reach

Enrollment
22

participants targeted

Target at P25-P50 for phase_1

Timeline
Completed

Started Aug 2010

Longer than P75 for phase_1

Geographic Reach
1 country

1 active site

Status
completed

Health score is calculated from publicly available data and should be used for screening purposes only.

Trial Relationships

Click on a node to explore related trials.

Study Timeline

Key milestones and dates

Study Start

First participant enrolled

August 1, 2010

Completed
1 month until next milestone

First Submitted

Initial submission to the registry

August 31, 2010

Completed
1 day until next milestone

First Posted

Study publicly available on registry

September 1, 2010

Completed
4 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

September 1, 2014

Completed
Same day until next milestone

Study Completion

Last participant's last visit for all outcomes

September 1, 2014

Completed
1.2 years until next milestone

Results Posted

Study results publicly available

November 11, 2015

Completed
Last Updated

May 22, 2017

Status Verified

April 1, 2017

Enrollment Period

4.1 years

First QC Date

August 31, 2010

Results QC Date

October 13, 2015

Last Update Submit

April 11, 2017

Conditions

Keywords

Aplastic AnemiaImmunosuppressionT-CellsHematopoiesisAutoimmunitySevere Aplastic Anemia

Outcome Measures

Primary Outcomes (1)

  • Blood Counts and Adverse Event Profile After 6 Months of Treatment.

    The safety endpoint will be toxicity profile after 6 months of treatment. The efficacy endpoint is complete response rate at 6 months, with complete response defined as blood counts no longer meeting the standard criteria for severe pancytopenia in severe aplastic anemia.

    6 months

Study Arms (1)

SAA hematologic response

EXPERIMENTAL

Treatment-naive severe aplastic anemia patients will receive a low dose of cyclophosphamide (120mg/kg) and low dose cyclosporine ( target therapeutic level of 100-200 micrograms per liter). Cyclophosphamide will be given once daily for 4 doses. Cyclosporine will be started after cyclophosphamide completion, cyclosporine will be given twice daily. The dosing will be modified to attain the therapeutic level.

Drug: CyclophosphamideDrug: Cyclosporine

Interventions

30 my/kg for 4 days

Also known as: Cytoxan
SAA hematologic response

daily to a trough of 100 t0 200 ng/ml

Also known as: Gengraf, Neoral, Sandimmune
SAA hematologic response

Eligibility Criteria

Age2 Years+
Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64), Older Adult (65+)

You may qualify if:

  • Severe aplastic anemia characterized by:
  • Bone marrow cellularity less than 30 percent (excluding lymphocytes)
  • AND
  • At least two of the following:
  • Absolute neutrophil count less than 500/ microL
  • Platelet count less than 20,000/ microL
  • Absolute reticulocyte count less than 60,000/ microL
  • Age greater than or equal to 2 years old
  • Weight greater than or equal to 12 kg

You may not qualify if:

  • Diagnosis of Fanconi anemia
  • Cardiac ejection fraction less than 30 percent (evaluated by ECHO)
  • Evidence of a clonal hematologic bone marrow disorder on cytogenetics. Patients with the presence of trisomy 8, loss of Y or del(20q) will not be excluded in the absence of dysplastic changes in the marrow. Patients with very severe neutropenia (ANC less than 200 /microL) will not be excluded initially if cytogenetics are not available or pending. If evidence of a clonal disorder is later identified, the patient will go off study.
  • Prior immunosuppressive therapy with high dose Cy or ATG
  • Infection not adequately controlled with appropriate therapy
  • Serologic evidence of HIV infection
  • Moribund status or concurrent hepatic, renal, cardiac, neurologic, pulmonary, infectious, or metabolic disease of such severity that it would preclude the patient s ability to tolerate protocol therapy, or that death within 30 days is likely
  • Subjects with cancer who are not considered cured, are on active chemotherapeutic treatment or who take drugs with hematological effects
  • Current pregnancy or unwillingness to take oral contraceptives or refrain from pregnancy if of childbearing potential
  • Not able to understand the investigational nature of the study or to give informed consent.

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (1)

National Institutes of Health Clinical Center, 9000 Rockville Pike

Bethesda, Maryland, 20892, United States

Location

Related Publications (5)

  • Rosenfeld S, Follmann D, Nunez O, Young NS. Antithymocyte globulin and cyclosporine for severe aplastic anemia: association between hematologic response and long-term outcome. JAMA. 2003 Mar 5;289(9):1130-5. doi: 10.1001/jama.289.9.1130.

    PMID: 12622583BACKGROUND
  • Young NS, Calado RT, Scheinberg P. Current concepts in the pathophysiology and treatment of aplastic anemia. Blood. 2006 Oct 15;108(8):2509-19. doi: 10.1182/blood-2006-03-010777. Epub 2006 Jun 15.

    PMID: 16778145BACKGROUND
  • Scheinberg P, Nunez O, Wu C, Young NS. Treatment of severe aplastic anaemia with combined immunosuppression: anti-thymocyte globulin, ciclosporin and mycophenolate mofetil. Br J Haematol. 2006 Jun;133(6):606-11. doi: 10.1111/j.1365-2141.2006.06085.x.

    PMID: 16704434BACKGROUND
  • Zaimoku Y, Patel BA, Adams SD, Shalhoub R, Groarke EM, Lee AAC, Kajigaya S, Feng X, Rios OJ, Eager H, Alemu L, Quinones Raffo D, Wu CO, Flegel WA, Young NS. HLA associations, somatic loss of HLA expression, and clinical outcomes in immune aplastic anemia. Blood. 2021 Dec 30;138(26):2799-2809. doi: 10.1182/blood.2021012895.

  • Scheinberg P, Townsley D, Dumitriu B, Scheinberg P, Weinstein B, Daphtary M, Rios O, Wu CO, Young NS. Moderate-dose cyclophosphamide for severe aplastic anemia has significant toxicity and does not prevent relapse and clonal evolution. Blood. 2014 Oct 30;124(18):2820-3. doi: 10.1182/blood-2014-05-573642. Epub 2014 Sep 3.

Related Links

MeSH Terms

Conditions

Anemia, AplasticNeutropeniaPancytopeniaAutoimmune Diseases

Interventions

CyclophosphamideCyclosporineCyclosporins

Condition Hierarchy (Ancestors)

AnemiaHematologic DiseasesHemic and Lymphatic DiseasesBone Marrow Failure DisordersBone Marrow DiseasesAgranulocytosisLeukopeniaCytopeniaLeukocyte DisordersImmune System Diseases

Intervention Hierarchy (Ancestors)

Phosphoramide MustardsNitrogen Mustard CompoundsMustard CompoundsHydrocarbons, HalogenatedHydrocarbonsOrganic ChemicalsPhosphoramidesOrganophosphorus CompoundsPeptides, CyclicMacrocyclic CompoundsPolycyclic CompoundsPeptidesAmino Acids, Peptides, and Proteins

Results Point of Contact

Title
Dr. Danielle Townsley
Organization
NIH NHLBI

Study Officials

  • Danielle M Townsley, M.D.

    National Heart, Lung, and Blood Institute (NHLBI)

    PRINCIPAL INVESTIGATOR

Publication Agreements

PI is Sponsor Employee
Yes

Study Design

Study Type
interventional
Phase
phase 1
Allocation
NA
Masking
NONE
Purpose
TREATMENT
Intervention Model
SINGLE GROUP
Sponsor Type
NIH
Responsible Party
PRINCIPAL INVESTIGATOR
PI Title
Hematology Clinician

Study Record Dates

First Submitted

August 31, 2010

First Posted

September 1, 2010

Study Start

August 1, 2010

Primary Completion

September 1, 2014

Study Completion

September 1, 2014

Last Updated

May 22, 2017

Results First Posted

November 11, 2015

Record last verified: 2017-04

Locations