Sirolimus and Cyclosporine for Treatment-Resistant Aplastic Anemia

NCT ID: NCT00319878

Last Updated: 2008-10-07

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

UNKNOWN

Clinical Phase

PHASE1/PHASE2

Total Enrollment

52 participants

Study Classification

INTERVENTIONAL

Study Start Date

2006-05-31

Study Completion Date

2009-12-31

Brief Summary

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Aplastic anemia is a rare autoimmune disorder in which the bone marrow production of blood cells is greatly decreased or absent. Symptoms include fatigue, weakness, tiny reddish-purple marks on the skin, abnormal bruising, and bleeding from the gums, nose, or intestine. While some cases of aplastic anemia are caused by medications, toxic exposures, or inherited genes, most often the cause remains unknown. The purpose of this study is to determine the safety and efficacy of combining two drugs, sirolimus and cyclosporine, for treating individuals with aplastic anemia that has not responded to other treatments.

Detailed Description

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The most successful treatment for aplastic anemia is bone marrow transplantation. However, few patients are eligible for this procedure. For others, treatment usually consists of immunosuppressive agents, such as antithymocyte globulin (ATG) and cyclosporine. Unfortunately, even with immunosuppressive therapy, relapse is common. New combinations of medications may offer alternative and more effective treatment options. Sirolimus and cyclosporine are two drugs routinely used to suppress the immune system and prevent rejection in patients who have received organ transplants. While cyclosporine has been proven effective for treating aplastic anemia, sirolimus has not been tested for this disease. This study will evaluate the safety and efficacy of sirolimus in combination with cyclosporine for treating individuals with aplastic anemia that has not responded to other treatments.

This study will last at least 6 months. Participants will first be screened to verify diagnosis of aplastic anemia. The screening will include a physical examination, blood test, bone marrow biopsy from the pelvic bone, and review of medications and medical history. Individuals who are eligible will then start the first treatment period. Participants will receive two medications: cyclosporine will be taken twice a day and sirolimus will be taken once a day. Depending on side effects, the doses of either drug may be temporarily stopped or lowered. On Day 1, blood will be drawn and females will undergo a pregnancy test. Subsequent study visits will occur weekly for the first month, every 2 weeks for 2 months, and then once a month for the remainder of the study. Each visit will include a physical examination, vital sign assessment, and review of side effects and medications. Blood tests will be performed weekly for the first 3 weeks, and then every 2 weeks.

After 6 months of treatment, if a participant has shown improvements in disease status without major side effects, the treatment will continue. Over time the doses may be lowered. If a participant has not improved while on the study medication, treatment will stop at 6 months. Whenever treatment is discontinued, the participant will again undergo a physical examination, blood tests, and bone marrow biopsy.

Conditions

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Anemia, Aplastic

Study Design

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Allocation Method

NON_RANDOMIZED

Intervention Model

SINGLE_GROUP

Primary Study Purpose

TREATMENT

Blinding Strategy

NONE

Study Groups

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1

Participants will be treated with sirolimus and cyclosporine. In phase I, each dose cohort will initially enroll three patients. If no dose-limiting toxicity (DLT) is observed by Day 28 in any patient of a cohort, then 3 patients will be treated with the next highest sirolimus dose. If 1 out of 3 patients in any cohort experiences a DLT, then 3 more patients will be enrolled in that cohort. If no more patients have a DLT by Day 28, then sirolimus dose escalation will proceed. If one or more patients experience a DLT then that dose level will be considered to be the maximum tolerated sirolimus dose, and Phase II patients will be treated at the next lowest level. Cyclosporine will be given as a twice daily oral dose.

Group Type EXPERIMENTAL

Sirolimus

Intervention Type DRUG

Oral loading dose followed by a once daily dose:

* Cohort 1: Loading Dose - 1.2 mg; Daily Dose - 0.4 mg
* Cohort 2: % Dose Increase - 100%; Loading Dose - 2.4 mg; Daily Dose - 0.8 mg
* Cohort 3: % Dose Increase - 67%; Loading Dose - 3.9 mg; Daily Dose - 1.3 mg
* Cohort 4: % Dose Increase - 50%; Loading Dose - 6.0 mg; Daily Dose - 2.0 mg

Cyclosporine

Intervention Type DRUG

Dose of 5 mg/kg divided as a twice daily oral dose

Interventions

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Sirolimus

Oral loading dose followed by a once daily dose:

* Cohort 1: Loading Dose - 1.2 mg; Daily Dose - 0.4 mg
* Cohort 2: % Dose Increase - 100%; Loading Dose - 2.4 mg; Daily Dose - 0.8 mg
* Cohort 3: % Dose Increase - 67%; Loading Dose - 3.9 mg; Daily Dose - 1.3 mg
* Cohort 4: % Dose Increase - 50%; Loading Dose - 6.0 mg; Daily Dose - 2.0 mg

Intervention Type DRUG

Cyclosporine

Dose of 5 mg/kg divided as a twice daily oral dose

Intervention Type DRUG

Eligibility Criteria

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Inclusion Criteria

* Diagnosis of moderate or severe aplastic anemia with bone marrow cellularity of less than 25%
* Falls within one of the following descriptions at the time of the original diagnosis:

1. For severe aplastic anemia, fulfills any two of the following three criteria: absolute neutrophil count less than 500/uL; absolute reticulocyte count less than 60,000/uL; and platelet count less than 20,000/uL
2. For moderate aplastic anemia, fulfills any two of the following three criteria: absolute neutrophil count less than 1200/ul; hemoglobin less than 8 g/dL with corrected reticulocyte count less than 1%; and platelet count less than 60,000/uL (Note: Participants who have progressed from moderate to severe aplastic anemia prior to study entry will be classified as having severe aplastic anemia)
* Diagnosis of refractory aplastic anemia, as defined by a failure to achieve at least a partial response to ATG within 6 months of treatment. Individuals who had a prior response to ATG but who have relapsed and not responded to salvage ATG are eligible. Individuals with relapsed disease who are not candidates for salvage ATG because they experienced a serious or life-threatening complication prior to ATG are also eligible.
* A Karnofsky performance status of at least 60%
* Adequate organ function, as defined by creatine levels less than 1.5 times the upper limit normal (ULN), and liver function tests (AST, bilirubin) less than 2 times the ULN
* Women of childbearing age must be willing to use effective contraception throughout the study

Exclusion Criteria

* Received ATG treatment less than 6 months prior to study entry
* Candidate for related allogeneic stem cell transplantation
* Active uncontrolled infection
* History of myelodysplastic syndrome or bone marrow cytogenetic abnormalities
* History of Fanconi's anemia or other congenital form of aplastic anemia
* Treatment with an investigational agent within 1 month of study entry
* HIV infection
* Pregnant or breastfeeding
Minimum Eligible Age

21 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Rare Diseases Clinical Research Network

NETWORK

Sponsor Role collaborator

Office of Rare Diseases (ORD)

NIH

Sponsor Role lead

Responsible Party

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UCLA Division of Hematology/Oncology

Locations

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UCLA Center for Health Sciences

Los Angeles, California, United States

Site Status RECRUITING

Lee Moffitt Cancer Center

Tampa, Florida, United States

Site Status RECRUITING

Taussig Cancer Center, Cleveland Clinic Foundation

Cleveland, Ohio, United States

Site Status RECRUITING

Penn State University Cancer Center

Hershey, Pennsylvania, United States

Site Status RECRUITING

Countries

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United States

Central Contacts

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Lynn Tihopu

Role: CONTACT

Phone: 310-794-0738

Meenal Chalukya

Role: CONTACT

Phone: 310-825-8091

Facility Contacts

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Ronald Paquette, MD

Role: primary

Alan List, MD

Role: primary

Jaroslaw P. Maciejewski, MD

Role: primary

Thomas Loughran, MD

Role: primary

References

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Maciejewski JP, Risitano AM. Aplastic anemia: management of adult patients. Hematology Am Soc Hematol Educ Program. 2005:110-7. doi: 10.1182/asheducation-2005.1.110.

Reference Type BACKGROUND
PMID: 16304367 (View on PubMed)

Young NS. Immunosuppressive treatment of acquired aplastic anemia and immune-mediated bone marrow failure syndromes. Int J Hematol. 2002 Feb;75(2):129-40. doi: 10.1007/BF02982017.

Reference Type BACKGROUND
PMID: 11939258 (View on PubMed)

Brodsky RA, Chen AR, Brodsky I, Jones RJ. High-dose cyclophosphamide as salvage therapy for severe aplastic anemia. Exp Hematol. 2004 May;32(5):435-40. doi: 10.1016/j.exphem.2004.02.002.

Reference Type BACKGROUND
PMID: 15145211 (View on PubMed)

Paquette RL. Diagnosis and management of aplastic anemia and myelodysplastic syndrome. Oncology (Williston Park). 2002 Sep;16(9 Suppl 10):153-61.

Reference Type BACKGROUND
PMID: 12380966 (View on PubMed)

Other Identifiers

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U54RR019397-01

Identifier Type: NIH

Identifier Source: secondary_id

View Link

BMF 5403

Identifier Type: -

Identifier Source: secondary_id

RDCRN 5403

Identifier Type: -

Identifier Source: org_study_id