Combination Chemotherapy Followed By Donor Stem Cell Transplant in Treating Patients With Hemophagocytic Lymphohistiocytosis

NCT ID: NCT00334672

Last Updated: 2013-09-17

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

PHASE3

Total Enrollment

288 participants

Study Classification

INTERVENTIONAL

Study Start Date

2006-03-31

Brief Summary

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RATIONALE: Drugs used in chemotherapy work in different ways to stop the growth of hemophagocytic lymphohistiocytosis cells, either by killing the cells or by stopping them from dividing. Giving more than one drug (combination chemotherapy) may kill more hemophagocytic lymphohistiocytosis cells. A donor stem cell transplant may be able to replace blood-forming cells that were destroyed by chemotherapy. Sometimes the transplanted cells from a donor can make an immune response against the body's normal cells. Cyclosporine and methotrexate may stop this from happening.

PURPOSE: This phase III trial is studying how well combination chemotherapy followed by a donor stem cell transplant works in treating patients with hemophagocytic lymphohistiocytosis.

Detailed Description

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OBJECTIVES:

Primary

* Provide and evaluate revised induction and maintenance therapy comprising etoposide, dexamethasone, and cyclosporine, in terms of achieving and maintaining an acceptable clinical condition in order to perform a curative allogeneic hematopoietic stem cell transplantation (AHSCT), in patients with primary inherited or severe and persistent secondary hemophagocytic lymphohistiocytosis (HLH).
* Evaluate and improve the outcome of AHSCT with various types of donors.
* Determine the prognostic importance of the state of remission at the time of AHSCT.
* Evaluate the neurological complications, in terms of early neurological alterations and cerebrospinal fluid (CSF) findings, in patients treated with this regimen.

Secondary

* Improve the understanding of the pathophysiology of HLH by conducting biological studies of genetics and cytotoxicity in these patients, including genotype-phenotype studies and the prognostic value of natural killer (NK) cell activity subtyping.

OUTLINE: This is a multicenter study.

* Induction therapy (weeks 1-8): Patients receive etoposide IV over 1-3 hours twice weekly in weeks 1 and 2 and then once weekly in weeks 3-8. Patients also receive dexamethasone IV or orally once daily and cyclosporine IV or orally twice daily in weeks 1-8. Patients with clinically evident, progressive neurological symptoms or an abnormal cerebrospinal fluid (CSF) (cell count and protein) that has not improved after 2 weeks of induction therapy undergo intrathecal therapy comprising methotrexate and hydrocortisone once weekly in weeks 3-6.

Patients are evaluated after 8 weeks of induction therapy. Patients with primary (i.e., familial) hemophagocytic lymphohistiocytosis (HLH) or genetic evidence of HLH proceed to maintenance therapy. Patients with severe and persistent secondary (i.e., nonfamilial) HLH and no genetic evidence of HLH proceed to maintenance therapy only if their disease is still active after induction therapy. Patients with nonfamilial HLH and no genetic evidence of HLH who have achieved complete remission (CR) discontinue treatment. If their disease reactivates, they may then proceed to allogeneic hematopoietic stem cell transplantation (AHSCT).

* Maintenance therapy (weeks 9-40): Patients receive dexamethasone IV on days 1-3 in weeks 10, 12, 14, 16, 18, 20, 22, 24, 26, 28, 30, 32, 34, 36, 38, and 40; etoposide IV over 1-3 hours once in weeks 9, 11, 13, 15, 17, 19, 21, 23, 25, 27, 29, 31, 33, 35, 37, and 39; and cyclosporine IV or orally twice daily in weeks 9-40.

After completion of maintenance therapy, patients with primary (i.e., familial) HLH, severe and persistent secondary (i.e., nonfamilial) HLH, or reactivating disease proceed to AHSCT. Patients with nonfamilial HLH who have completed maintenance therapy, but do not go on to receive AHSCT, may be recommended for additional maintenance therapy at the discretion of the treating physician.

* AHSCT:

* Preparative regimen: Patients receive a preparative regimen comprising busulfan orally or IV four times daily on days -8 to -5, etoposide IV over 6 hours on day -4, and cyclophosphamide IV over 1 hour on days -3 and -2. Patients who are undergoing unrelated AHSCT, also receive antithymocyte globulin (ATG) IV over 12 hours on days -3 to -1.
* Transplantation: Patients undergo AHSCT on day 0.
* Graft-versus-host disease prophylaxis: Beginning on day -1, patients receive cyclosporine IV continuously and then orally, when tolerated, once daily for 6-12 months. Patients also receive methotrexate\* IV on days 1, 3, and 6.

NOTE: \*As a substitute for methotrexate, patients may receive oral mycophenolate mofetil twice daily on days 0-40, followed by a taper and discontinuation.

Patients undergo periodic blood collection and bone marrow biopsies for biological studies.

After completion of study treatment, patients are followed periodically for up to 5 years.

PROJECTED ACCRUAL: A total of 288 patients will be accrued for this study.

Conditions

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Nonneoplastic Condition

Keywords

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hemophagocytic lymphohistiocytosis

Study Design

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Primary Study Purpose

TREATMENT

Blinding Strategy

NONE

Interventions

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anti-thymocyte globulin

Intervention Type BIOLOGICAL

busulfan

Intervention Type DRUG

cyclophosphamide

Intervention Type DRUG

cyclosporine

Intervention Type DRUG

dexamethasone

Intervention Type DRUG

etoposide

Intervention Type DRUG

methotrexate

Intervention Type DRUG

mycophenolate mofetil

Intervention Type DRUG

therapeutic hydrocortisone

Intervention Type DRUG

laboratory biomarker analysis

Intervention Type OTHER

allogeneic hematopoietic stem cell transplantation

Intervention Type PROCEDURE

biopsy

Intervention Type PROCEDURE

Eligibility Criteria

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Inclusion Criteria

* Primary HLH (i.e., familial hemophagocytic lymphohistiocytosis \[FLH\]) OR secondary HLH (i.e., severe acquired form of HLH)
* Acceptable donor meeting 1 of the following criteria:

* HLA-identical related donor
* Matched unrelated donor
* Mismatched unrelated donor
* Familial haploidentical donor

PATIENT CHARACTERISTICS:

* Not specified

PRIOR CONCURRENT THERAPY:

* No prior cytotoxic treatment for HLH
* No prior cyclosporine treatment for HLH
Maximum Eligible Age

17 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Children's Cancer and Leukaemia Group

OTHER

Sponsor Role lead

Principal Investigators

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Vasanta Nanduri, MD

Role: STUDY_CHAIR

Watford General Hospital

Locations

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Birmingham Children's Hospital

Birmingham, England, United Kingdom

Site Status

Institute of Child Health at University of Bristol

Bristol, England, United Kingdom

Site Status

Addenbrooke's Hospital

Cambridge, England, United Kingdom

Site Status

Watford General Hospital

Herts, England, United Kingdom

Site Status

Leeds Cancer Centre at St. James's University Hospital

Leeds, England, United Kingdom

Site Status

Royal Liverpool Children's Hospital, Alder Hey

Liverpool, England, United Kingdom

Site Status

Great Ormond Street Hospital for Children

London, England, United Kingdom

Site Status

Royal Manchester Children's Hospital

Manchester, England, United Kingdom

Site Status

Children's Hospital - Sheffield

Sheffield, England, United Kingdom

Site Status

Southampton General Hospital

Southampton, England, United Kingdom

Site Status

Royal Aberdeen Children's Hospital

Aberdeen, Scotland, United Kingdom

Site Status

Royal Hospital for Sick Children

Edinburgh, Scotland, United Kingdom

Site Status

Royal Hospital for Sick Children

Glasgow, Scotland, United Kingdom

Site Status

Countries

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Ireland United Kingdom

Other Identifiers

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CCLG-LCH-2006-02

Identifier Type: -

Identifier Source: secondary_id

EU-20619

Identifier Type: -

Identifier Source: secondary_id

UKCCSG-HLH-2004

Identifier Type: -

Identifier Source: secondary_id

EUDRACT-2005-002187-28

Identifier Type: -

Identifier Source: secondary_id

CDR0000481605

Identifier Type: -

Identifier Source: org_study_id