NCT00426101

Brief Summary

Without therapy HLH is often fatal, and often rapidly fatal. The treatment protocol HLH-94 has improved survival markedly as compared to the survival earlier. We now aim to improve survival further.

Trial Health

87
On Track

Trial Health Score

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

Enrollment
368

participants targeted

Target at P50-P75 for phase_3

Timeline
Completed

Started Jan 2004

Longer than P75 for phase_3

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

January 1, 2004

Completed
3.1 years until next milestone

First Submitted

Initial submission to the registry

January 23, 2007

Completed
1 day until next milestone

First Posted

Study publicly available on registry

January 24, 2007

Completed
4.9 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

December 1, 2011

Completed
6 years until next milestone

Study Completion

Last participant's last visit for all outcomes

December 1, 2017

Completed
Last Updated

July 5, 2018

Status Verified

July 1, 2018

Enrollment Period

7.9 years

First QC Date

January 23, 2007

Last Update Submit

July 2, 2018

Conditions

Keywords

Hemophagocytic lymphohistiocytosis

Outcome Measures

Primary Outcomes (1)

  • Survival

    1-year after diagnosis

Secondary Outcomes (1)

  • Late effects

    5-years after diagnosis

Study Arms (1)

Etoposide, Dexamethasone, Cyclosporin A plus IT MTX & Steroids

EXPERIMENTAL

As compared to the HLH-94 treatment, the main changes are that 1. Cyclosporin A is administered from day 1 and 2. Intrathecal steroids are added to the intrathecal methotrexate. Drugs, dosage, frequency and duration are described in the paragraph "Interventions" below.

Drug: DexamethasoneDrug: EtoposideDrug: CyclosporinProcedure: Intrathecal therapyProcedure: Stem cell transplant

Interventions

10 mg/m2 daily wk 1-2 5 mg/m2 daily wk 3-4 2.5 mg/m2 daily wk 5-6 1.25 mg/m2 daily wk 7 Steroids tapered wk 8 If continuation: Pulses every 2nd wk, 10 mg/m2 for 3 days

Etoposide, Dexamethasone, Cyclosporin A plus IT MTX & Steroids

150 mg/m2 iv twice/wk (wk 1-2) 150 mg/m2 iv once/wk (wk 3-8) If continuation: 150 mg/m2 iv, every 2nd wk

Etoposide, Dexamethasone, Cyclosporin A plus IT MTX & Steroids

WK 1-8: \- Aim at around 200 microgram/L (trough value). Start: 6 mg/kg daily (divided in 2 daily doses) wk 1, if kidney function is normal. If continuation: \- Aim for around 200 microgram/L. Monitor GFR.

Etoposide, Dexamethasone, Cyclosporin A plus IT MTX & Steroids

If at 2 wks there are progressive neurological symptoms or if an abnormal CSF (cell count and protein) has not improved, then give 4 wkly intrathecal inj. Be aware that some pat may have increased intracranial pressure. Methotrexate: \<1 yr 6 mg, 1-2 yrs 8 mg, 2-3 yrs 10 mg, \>3 yrs 12 mg. Prednisolone: \<1 yr 4 mg, 1-2 yrs 6 mg, 2-3 yrs 8 mg, \>3 yrs 10 mg.

Etoposide, Dexamethasone, Cyclosporin A plus IT MTX & Steroids

The SCT procedure is up to the treating physician. However, a suggested regimen is provided.

Etoposide, Dexamethasone, Cyclosporin A plus IT MTX & Steroids

Eligibility Criteria

AgeUp to 18 Years
Sexall
Healthy VolunteersNo
Age GroupsChild (0-17), Adult (18-64)

You may qualify if:

  • Patients who fulfil the diagnostic criteria of HLH.

You may not qualify if:

  • Prior cytotoxic or cyclosporin treatment for HLH.

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (1)

Childhood Cancer Research Unit, Karolinska Hospital

Stockholm, S-171 76, Sweden

Location

Related Publications (3)

  • Henter JI, Horne A, Arico M, Egeler RM, Filipovich AH, Imashuku S, Ladisch S, McClain K, Webb D, Winiarski J, Janka G. HLH-2004: Diagnostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis. Pediatr Blood Cancer. 2007 Feb;48(2):124-31. doi: 10.1002/pbc.21039.

    PMID: 16937360BACKGROUND
  • Bergsten E, Horne A, Arico M, Astigarraga I, Egeler RM, Filipovich AH, Ishii E, Janka G, Ladisch S, Lehmberg K, McClain KL, Minkov M, Montgomery S, Nanduri V, Rosso D, Henter JI. Confirmed efficacy of etoposide and dexamethasone in HLH treatment: long-term results of the cooperative HLH-2004 study. Blood. 2017 Dec 21;130(25):2728-2738. doi: 10.1182/blood-2017-06-788349. Epub 2017 Sep 21.

  • Bergsten E, Horne A, Hed Myrberg I, Arico M, Astigarraga I, Ishii E, Janka G, Ladisch S, Lehmberg K, McClain KL, Minkov M, Nanduri V, Rosso DA, Sieni E, Winiarski J, Henter JI. Stem cell transplantation for children with hemophagocytic lymphohistiocytosis: results from the HLH-2004 study. Blood Adv. 2020 Aug 11;4(15):3754-3766. doi: 10.1182/bloodadvances.2020002101.

MeSH Terms

Conditions

Lymphohistiocytosis, Hemophagocytic

Interventions

DexamethasoneEtoposideCyclosporineStem Cell Transplantation

Condition Hierarchy (Ancestors)

Histiocytosis, Non-Langerhans-CellHistiocytosisLymphatic DiseasesHemic and Lymphatic Diseases

Intervention Hierarchy (Ancestors)

PregnadienetriolsPregnadienesPregnanesSteroidsFused-Ring CompoundsPolycyclic CompoundsSteroids, FluorinatedPodophyllotoxinTetrahydronaphthalenesNaphthalenesPolycyclic Aromatic HydrocarbonsHydrocarbons, AromaticHydrocarbons, CyclicHydrocarbonsOrganic ChemicalsGlucosidesGlycosidesCarbohydratesCyclosporinsPeptides, CyclicMacrocyclic CompoundsPeptidesAmino Acids, Peptides, and ProteinsCell TransplantationCell- and Tissue-Based TherapyBiological TherapyTherapeuticsTransplantationSurgical Procedures, Operative

Study Officials

  • Jan-Inge Henter, MD, PhD

    Karolinska Institutet

    PRINCIPAL INVESTIGATOR

Study Design

Study Type
interventional
Phase
phase 3
Allocation
NA
Masking
NONE
Purpose
TREATMENT
Intervention Model
SINGLE GROUP
Sponsor Type
OTHER
Responsible Party
PRINCIPAL INVESTIGATOR
PI Title
Professor

Study Record Dates

First Submitted

January 23, 2007

First Posted

January 24, 2007

Study Start

January 1, 2004

Primary Completion

December 1, 2011

Study Completion

December 1, 2017

Last Updated

July 5, 2018

Record last verified: 2018-07

Locations