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Phase 2 Completed N=33 Treatment

Pharmacokinetics of Oral Hydroxyurea Solution

Sickle Cell Disease · Sickle-Cell; Hemoglobin Disease, Thalassemia · Sickle Cell-beta-thalassemia · Sickle Cell Hemoglobin C
Source: ClinicalTrials.gov NCT03763656 ↗
Enrolled (actual)
33
Serious AEs
18.8%
Results posted
Oct 2024
Primary outcomePrimary: Clearance (CL/F) — 3.61 L/hr

Summary

An open label, safety and pharmacokinetic study of oral hydroxyurea solution administered to children from 6 months to 17.99 years (i.e. to the day before 18th birthday), with a 12 to 15 month treatment period for each participant. The study treatment duration will be for 6 months at the maximum tolerated dose [MTD], which is usually reached by 6 months after initiation of treatment. For patients in whom time to MTD is longer than 6 months or not achieved at all, the maximum duration of study treatment will be 15 months.

Outcome Measures

OutcomeResultp-value
PRIMARY
Clearance (CL/F)
3.61
PRIMARY
Volume of Distribution (V/F)
11.4
PRIMARY
Time to Maximum Concentration (Tmax)
1.253; 1.265; 1.238; 1.269
PRIMARY
Maximum Plasma Concentration Cmax (ug/mL)
13.124; 12.807; 13.068; 13.404
PRIMARY
Area Under Plasma Concentration Time Curve (AUC 0-Inf)
64.721; 62.537; 62.899; 68.948
PRIMARY
Terminal Half-life (Hours)
3.880; 4.107; 3.949; 3.634
SECONDARY
Adverse Events
339; 339; 7; 19; 28; 53
SECONDARY
Absolute Neutrophil Count (ANC)
4.90; 4.30; 5.38; 4.43; 2.70; 2.27
SECONDARY
White Blood Cell Count (Leukocytes)
13.01; 12.24; 14.59; 10.88; 7.48; 7.90
SECONDARY
Platelets
409.06; 383.80; 381.31; 466.10; 315.10; 333.67
SECONDARY
Mean Corpuscular Hemoglobin (MCH)
27.57; 23.80; 28.36; 28.53; 33.36; 28.95
SECONDARY
Hematocrit
25.02; 30.60; 23.24; 24.30; 27.74; 30.38
SECONDARY
Bilirubin
37.43; 14.78; 39.60; 47.53; 26.12; 7.42
SECONDARY
Elevation in Liver Function Tests (LFTs)
3.55; 4.19; 3.60; 3.10; 3.57; 4.06
SECONDARY
Hemoglobin
8.32; 9.76; 8.07; 7.99; 9.47; 10.60
SECONDARY
Bacterial Infections
7; 0; 3; 4; 17; 1
SECONDARY
Viral Infections
8; 0; 8; 0; 23; 4
SECONDARY
Fungal Infections
1; 0; 0; 1; 4; 1
SECONDARY
Leg Ulcers
0; 0; 0; 0; 1; 0
SECONDARY
Fetal Hemoglobin
11.90; 21.52; 10.94; 5.57; 23.53; 26.32
SECONDARY
Mean Corpuscular Volume (MCV)
83.13; 76.42; 83.25; 86.30; 97.64; 81.68
SECONDARY
Cystatin C
7.52; 8.89; 6.87; 7.18; 7.50; 8.36
SECONDARY
Incidence of Acute Vaso-Occlusive Pain Crises (VOC)
0.5; 0.0; 0.7; 0.6; 0.2; 0.3
SECONDARY
Number and Frequency of Blood Transfusions
30; 21; 9
SECONDARY
Acute Chest Syndrome (ACS)
0.5; 0.00; 0.6; 0.7; 0.2; 0.00
SECONDARY
Hospitalizations
1.7; 0.3; 2.0; 1.9; 0.3; 0.3
SECONDARY
Dose Escalation i.e. Maximum Tolerated Dose (MTD)
25.63; 23.50; 26.11; 26.67
SECONDARY
Other SCA-related Hospitalizations
0.4; 0.3; 0.4; 0.4; 0.0; 0.00
SECONDARY
Parent/Caregiver Palatability and Acceptability Questionnaire
84.2; 71.3; 91.1; 80.8; 83.8; 74.8
SECONDARY
Vitamin D
59.35; 83.07; 65.43; 35.39; 62.26; 86.44
SECONDARY
Other Non-SCA-related Hospitalizations
0.2; 0.00; 0.3; 0.2; 0.00; 0.00

Eligibility Criteria

Inclusion Criteria

  • Male or female aged from 6 months to 17.99 years of age (i.e. to the day before 18th birthday).
  • Diagnosis of sickle cell anemia (HbSS and HbSβº).
  • Parent(s)/legal guardian able and willing to provide written informed consent for the child to take part in the study.
  • Where applicable, the child should assent to undergo blood sampling for pharmacokinetic and biochemistry purposes and to allow physiological measurements to be made.

Exclusion Criteria

  • Any clinically significant medical condition or abnormality, which, in the opinion of the Investigator, might have compromised the safety of the patient or which might have interfered with the study.
  • Hydroxyurea use within 6 months before enrolment.
  • Renal insufficiency (known creatinine more than twice the upper limit of normal (ULN) for age and >1.0 mg/dL [88.4 μmol/L]).
  • Clinical evidence of hepatic compromise with alanine aminotransferase (ALT) >3 times the ULN (a temporary swing in ALT did not result in exclusion).
  • Other significant organ system dysfunction based on the site Investigators discretion.
  • Severe active infections: fungal, viral or bacterial (as confirmed by culture), examples included tuberculosis, malaria, active hepatitis, osteomyelitis or any other illness that would have precluded the use of HU in normal clinical practice.
  • Active chronic leg ulcers.
  • Known allergy to oral HU solution or any of the excipients.
  • Positive pregnancy test for females of child-bearing potential (in post-menarcheal females) before initiation of treatment, unless participant was sexually abstinent. Note: True abstinence was considered as being in line with the preferred and usual lifestyle of the participant. Periodic abstinence (such as calendar, ovulation, symptothermal, post-ovulation methods) and withdrawal are not acceptable methods of contraception.
  • Inadequate contraception measures in sexually active females (post-menarcheal females) and males of child-bearing age (see Section 9.5.1.10.4).
  • Breastfeeding at study initiation.
  • Participation in another clinical trial of an IMP.
  • Known infection with HIV.
View full record on ClinicalTrials.gov →

Data sourced from ClinicalTrials.gov (NCT03763656). 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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