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Phase 2 Completed N=117 Treatment

Study of Dose Confirmation and Safety of Crizanlizumab in Pediatric Sickle Cell Disease Patients

Sickle Cell Disease (SCD)
Source: ClinicalTrials.gov NCT03474965 ↗
Enrolled (actual)
117
Serious AEs
50.4%
Results posted
Jul 2025
Primary outcomePrimary: Pharmacokinetics (PK): AUCd15 of Crizanlizumab After First Dose - Part A — 10500; 8180; 20600; 14400 hr*ug/mL

Summary

The purpose of this study was to confirm and to establish appropriate dosing and to evaluate the safety in pediatric participants ages 2 to <18 years with a history of Vaso-Occlusive Crisis (VOC) with or without Hydroxyurea/Hydroxycarbamide (HU/HC), receiving crizanlizumab for 2 years. The efficacy and safety of crizanlizumab was previously demonstrated in adults with sickle cell disease. The approach was to extrapolate from the pharmacokinetics (PK)/pharmacodynamics (PD) already established in the adult population. The study was designed as a Phase II, multicenter, open-label study.

Outcome Measures

OutcomeResultp-value
PRIMARY
Pharmacokinetics (PK): AUCd15 of Crizanlizumab After First Dose - Part A
10500; 8180; 20600; 14400
PRIMARY
Pharmacokinetics (PK) - AUCtau for Serum Crizanlizumab After Multiple Doses - Part A - Steady State
15800; 14800; 35700; 22100
PRIMARY
Pharmacokinetics (PK) - Cmax for Crizanlizumab After First Dose and Multiple Doses - Part A - Steady State
80.5; 65.9; 175; 110; 95.6; 77.5
PRIMARY
Pharmacodynamics (PD) - P-selectin Inhibition Parameters for Crizanlizumab After First Dose - Part A - AUCd15
33700; 34400; 33200
PRIMARY
Pharmacodynamics (PD) - P-selectin Inhibition Parameters for Crizanlizumab - Part A - AUCtau After Multiple Dose - Steady State
66700; 64800; 68600; 66300
PRIMARY
Frequency of Any Adverse Events (AEs) as a Measure of Safety and Tolerability
47; 13; 38; 14; 16; 5
SECONDARY
Annualized Rate Vaso Occlusive Crisis (VOC) Events Leading to Healthcare Visit in Clinic / Emergency Room (ER) / Hospital
3.00; 1.00; 2.00; 1.00; 1.80; 0.79
SECONDARY
Annualized Rate Vaso Occlusive Crisis (VOC) Events Treated at Home (Based on Documentation by Health Care Provider Following Phone Contact With the Patient)
0.94; 2.71; 0.49; 0.73
SECONDARY
Annualized Rate Vaso Occlusive Crisis (VOC) Events Leading to Healthcare Visit - Uncomplicated Pain Crisis
1.79; 0.98; 1.47; 0.97
SECONDARY
Annualized Rate Vaso Occlusive Crisis (VOC) Events Leading to Healthcare Visit - Acute Chest Syndrome
0.49; 0.64; 0.49; 0.49
SECONDARY
Annualized Rate Vaso Occlusive Crisis (VOC) Events Treated at Home (Based on Documentation by Health Care Provider Following Phone Contact With the Patient) - Hepatic Sequestration
SECONDARY
Annualized Rate Vaso Occlusive Crisis (VOC) Events Leading to Healthcare Visit - Splenic Sequestration
0.00; 0.00; 0.00
SECONDARY
Annualized Rate Vaso Occlusive Crisis (VOC) Events Leading to Healthcare Visit - Priapism
0.49; 0.00
SECONDARY
Annualized Rate of Hospitalizations and Emergency Room (ER) Visits (VOC-related)
1.78; 0.50; 1.01; 0.74; 2.00; 0.00
SECONDARY
Annualized Rate of Hospitalizations and Emergency Room (ER) Visits (Total)
2.68; 1.46; 1.59; 1.74; 2.00; 1.00
SECONDARY
Annualized Days of Emergency Room (ER) / Hospitalization (Both Overall and VOC-related)
6.74; 0.98; 5.36; 2.20; 12.37; 5.41
SECONDARY
Annualized Rate of Dactylitis Events
SECONDARY
Absolute Change From Baseline in Hemoglobin
-1.26; 2.33; -0.33; 1.55; -5.38; 2.33
SECONDARY
Immunogenicity: Measurement of Anti-drug Antibodies (ADA) to Crizanlizumab
50; 13; 39; 14; 0; 0
SECONDARY
Notable On-treatment Findings From the Electrocardiogram (ECG) Assessments
1; 1; 2; 1; 0; 0
SECONDARY
Growth and Sexual Maturation Assessments (Tanner Stage) - Abnormalities - for Female Participants at Risk of Delayed Puberty at Start Date of Study Treatment
0; 0; 0; 0; 0; 0
SECONDARY
Growth and Sexual Maturation Assessments (Tanner Stage) - Abnormalities - for Male Participants at Risk of Delayed Puberty at Start Date of Study Treatment
0; 0; 0; 0; 4; 0
SECONDARY
PK Pre-dose Concentrations of Crizanlizumab Prior to Each Study Drug Dose - Part A
16.8; 14.1; 30.1; 19.8; 10.7; 8.70
SECONDARY
PK Pre-dose Concentrations of Crizanlizumab Prior to Each Study Drug Dose - Parts A and B
16.6; 14.1; 23.6; 20.0; 10.3; 8.70
SECONDARY
Percent P-selectin Inhibition of Crizanlizumab Prior to Dosing - Part A
100; 97.9; 99.1; 98.6; 99.0; 94.2
SECONDARY
Percent P-selectin Inhibition of Crizanlizumab Prior to Dosing - Part A and B
99.3; 97.9; 98.3; 97.6; 93.7; 94.6
SECONDARY
Adverse Events by Preferred Term Related to Study Treatment
16; 5; 14; 4; 5; 2

Eligibility Criteria

Inclusion Criteria

  • Male or female patients ages 2 to 10 years of age, and Lansky ≥ 50 for patients ≤ 10 years of age.
  • Patient must meet the following laboratory values prior to Week 1 Day 1: Absolute Neutrophil Count ≥1.0 x 109/L , Platelets ≥75 x 109/L, Hemoglobin (Hgb) > 5.5 g/dL
  • Patient must have adequate renal and hepatic function as defined:Estimated Glomerular filtration rate (eGFR) ≥ 75 mL/min/1.73 m2 using Schwartz formula, Direct (conjugated) bilirubin ≤ 2.0 x ULN, Alanine transaminase (ALT) ≤ 3.0 x ULN,
  • Transcranial Doppler (TCD) for patients aged 2 to < 16 years at time of screening, with HbSS, HbSβ0-thalassemia, and HbSD disease indicating low risk for stroke (per investigator). Please refer to Section 7.2.2.6 for details
  • Written informed consent/assent, according to local guidelines, signed by the patient and / or by the parents or legal guardian prior to any study related screening procedures are performed.
  • Female of non-childbearing potential or with negative serum pregnancy test on Screening and a negative urine pregnancy test (dipstick) prior to dosing on Day 1.

Exclusion Criteria

  • History of stem cell transplant.
  • Received any blood products within 30 days prior to Week 1 Day 1 dosing.
  • Plan to participate in a chronic transfusion program (pre-planned series of transfusions for prophylactic purposes) or undergo exchange transfusions/plasmapheresis during the study. Patients requiring episodic transfusion (simple or exchange) in response to worsened anemia or VOC are permitted.
  • Patients with bleeding disorders

6.Contraindication or hypersensitivity to any drug from similar class as study drug or to any excipients of the study drug formulation.

7.History of severe hypersensitivity reaction to other monoclonal antibodies, which in the opinion of the investigator may pose an increased risk of serious infusion reaction 8.Received a monoclonal antibody or immunoglobulin-based therapy within 6 months of Screening, or has documented immunogenicity to a prior monoclonal antibody.

9.Received active treatment on another investigational trial within 30 days (or 5 half -lives of that agent, whichever is greater) prior to Screening or plans to participate in another investigational drug trial.

10.Pregnant females or females who have given birth within the past 90 days or who are breastfeeding.

  • Any documented history of a clinical stroke or intracranial hemorrhage, or an uninvestigated neurologic finding within the past 12 months. Silent infarcts (only present on imaging) are not excluding patients from study participation 12.Any abnormal TCD within the past 12 months. 13.Use of therapeutic anticoagulation (prophylactic doses permitted) or antiplatelet therapy (other than aspirin) within the 10 days prior to Week 1 Day 1 dosing.

14.Hospitalized within 7 days prior to Week 1 Day 1 dosing. 15.Planning to undergo a major surgical procedure during the duration of the study.

16.Planning to initiate or terminate HU/HC or L-glutamine while on study (except if needed to terminate for safety reasons).

17.Patient with active human immunodeficiency virus (HIV) infection (detectable viral load).

18.Patients with known active Hepatitis B infection. 19.Patients with known Hepatitis C history. 20.Significant active infection or immune deficiency (including chronic use of immunosuppressive drugs) in the opinion of the investigator.

21.Malignant disease. Exceptions to this exclusion include the following: malignancies that were treated curatively and have not recurred within 2 years prior to study treatment; any completely resected carcinoma in situ.

22.Has a serious mental or physical illness, which, in the opinion of the Investigator would compromise participation in the study.

23.Any condition which, in the opinion of the investigator, is likely to interfere with the successful collection of the measurements required for the study 24.Resting QTcF ≥450 msec at pretreatment (baseline) for p

View full record on ClinicalTrials.gov →

Data sourced from ClinicalTrials.gov (NCT03474965). Outcome figures and adverse-event rates are extracted automatically from the registry's posted results and are provided for clinician reference, not as a substitute for the primary publication. Informational only — not medical advice.

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