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A Study to Evaluate the Efficacy, Safety and Pharmacokinetics of Emapalumab in Adult Patients With HLH

Phase 2
Terminated
Conditions
Hemophagocytic Lymphohistiocytoses
Interventions
Registration Number
NCT03985423
Lead Sponsor
Swedish Orphan Biovitrum
Brief Summary

Hemophagocytic lymphohistiocytosis (HLH) is a rare, life-threatening condition characterized by uncontrolled hyperinflammation which may develop on the background of several clinical conditions (e.g. autoimmune disease, infection, malignancy). Emapalumab (previously referred to as NI-0501) is a monoclonal antibody neutralizing interferon-gamma (IFN-gamma), a key cytokine driving the inflammation and tissue damage seen in HLH. The purpose of this study is to assess the efficacy, safety and pharmacokinetics of emapalumab in adult patients with secondary HLH.

Detailed Description

Study NI-0501-10 is an open-label, single arm, multicenter, Phase 2/3 interventional study.

The study enrolls adult patients with hemophagocytic lymphohistiocytosis (HLH), specifically newly diagnosed patients with malignancy-associated HLH (M-HLH), and newly diagnosed or previously treated patients with non-malignancy-associated HLH.

Recruitment & Eligibility

Status
TERMINATED
Sex
All
Target Recruitment
7
Inclusion Criteria
  • Male and female patients of age 18 and older at the time of HLH diagnosis
  • Fulfilment of 5 of the 8 HLH-2004 clinical criteria
  • Patients diagnosed with malignancy-associated HLH must be treatment naïve; patients diagnosed with HLH driven by any other etiology or idiopathic can be either treatment naïve or treatment experienced
  • Patients with non-malignancy-associated or idiopathic HLH who have already received conventional therapy for HLH must have failed prior treatment as per the treating physician's judgement
  • Informed consent signed by the patient or by the patient's legally authorized representative(s) (as required by local law)
  • Willing to use highly effective methods of contraception from study drug initiation to 6 months after the last dose of study drug, if female and of childbearing potential.
Exclusion Criteria
  • Primary HLH
  • Current or scheduled administration of therapies known to trigger the cytokine release syndrome (e.g. chimeric antigen receptor (CAR)-modified T cells, bispecific T cell-engaging antibodies)
  • Current or scheduled administration of PD-1/PD-L1/CTLA-4 inhibitors
  • Life-expectancy associated with the underlying disease (triggering HLH) < 3 months
  • Ongoing participation in an investigational trial, or administration of any investigational treatment within 30 days
  • History of hypersensitivity or allergy to any components of emapalumab
  • Active mycobacteria, Histoplasma capsulatum, or Leishmania infections
  • Evidence of latent tuberculosis
  • Receipt of a bacille Calmette-Guerin (BCG) vaccine within 12 weeks prior to Screening
  • Receipt of a live or attenuated live (other than BCG) vaccine within 6 weeks prior to Screening

Study & Design

Study Type
INTERVENTIONAL
Study Design
SINGLE_GROUP
Arm && Interventions
GroupInterventionDescription
EmapalumabEmapalumab-LzsgPatients were administered Emapalumab-Lzsg by intravenous (i.v.) infusion over a period of 1 to 2 hours, at an initial dose of 6 mg/kg and continued at 3 mg/kg, every 3 days for the first 2 weeks (Study Day \[SD\] 15), and then twice-a-week. If the treating physician deemed appropriate, the dose of emapalumab could be increased (up to 10 mg/kg), guided by clinical and laboratory response.
Primary Outcome Measures
NameTimeMethod
Overall ResponseWeek 4

Achievement of either a Complete or Partial Response

Complete Response is adjudicated if the following are observed:

* No fever = body temperature \<37.5°C

* Normal spleen size

* No cytopenia = Absolute Neutrophil Counts \>=1.0x10\^9/L and platelet count \>=100x10\^9/L \[absence of G-CSF and transfusion support must be documented for at least 4 days to report no cytopenia\]

* No hyperferritinemia = serum level is \<2000 µg/L

* No evidence of coagulopathy, i.e., normal D-Dimer and/or normal (\>150 mg/dL) fibrinogen levels

* No neurological and CSF abnormalities attributed to HLH

* No sustained worsening of sCD25 (as indicated by at least two consecutive measurements that are \>2-fold higher than baseline)

Partial Response is adjudicated if there is an improvement (\>50% change from baseline or normalization) of at least 3 HLH clinical and laboratory criteria (including Central Nervous System abnormalities).

Secondary Outcome Measures
NameTimeMethod
Duration of ResponseUp to 1 year after last emapalumab administration

As no data are reported for this outcome measure, additional method is not applicable in the outcome measure description.

Best Response on TreatmentWeek 4; End of Treatment Visit (on average of 12 weeks)

As no data are reported for this outcome measure, additional method is not applicable in the outcome measure description.

Overall ResponseEnd of Treatment Visit (on average of 12 weeks)

As no data are reported for this outcome measure, additional method is not applicable in the outcome measure description.

Hemophagocytic Lymphohistiocytosis RelapseUp to 1 year after last emapalumab administration

As no data are reported for this outcome measure, additional method is not applicable in the outcome measure description.

Serum Concentrations of EmapalumabUp to 1 year after last emapalumab administration

As no data are reported for this outcome measure, additional method is not applicable in the outcome measure description.

Overall SurvivalEnd of Treatment Visit (on average of 12 weeks)

As no data are reported for this outcome measure, additional method is not applicable in the outcome measure description.

Time to Complete Response or Partial ResponseWeek 4; End of Treatment visit (on average of 12 weeks)

As no data are reported for this outcome measure, additional method is not applicable in the outcome measure description.

Incidence, Severity, Causality and Outcomes of Serious Adverse Events and Non-serious Adverse EventsUp to 1 year after last emapalumab administration

As no data are reported for this outcome measure, additional method is not applicable in the outcome measure description.

Serum Biomarker LevelsUp to 1 year after last emapalumab administration

Levels of interferon-gamma, C-X-C chemokine ligand 9, soluble CD25, interleukin-6.

Incidence of Anti-Drug Antibodies Against EmapalumabUp to 1 year after last emapalumab administration

As no data are reported for this outcome measure, additional method is not applicable in the outcome measure description.

Trial Locations

Locations (1)

MD Anderson Cancer Center

🇺🇸

Houston, Texas, United States

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