Trial Outcomes & Findings for Prospective Biomarkers of Bone Metabolism in Hemophilia A (NCT NCT02306694)

NCT ID: NCT02306694

Last Updated: 2020-04-01

Results Overview

BMD was measured as Z-scores/T-scores using Dual-Energy X-ray Absorptiometry (DEXA) scanning; specifically looking at Spine, Hip/Neck, and Hip total scores. BMD Z-scores compare what would be expected in someone your age and body size. A Z-score, is a unit of standard deviation, where above 0 would indicate the bones are more dense than expected, while a Z-score below 0 would indicate the bones were less dense.

Recruitment status

COMPLETED

Study phase

PHASE3

Target enrollment

16 participants

Primary outcome timeframe

5 days

Results posted on

2020-04-01

Participant Flow

Participant milestones

Participant milestones
Measure
Open Label
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Overall Study
STARTED
16
Overall Study
COMPLETED
12
Overall Study
NOT COMPLETED
4

Reasons for withdrawal

Reasons for withdrawal
Measure
Open Label
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Overall Study
Withdrawal by Subject
3
Overall Study
Lost to Follow-up
1

Baseline Characteristics

Prospective Biomarkers of Bone Metabolism in Hemophilia A

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
Open Label
n=16 Participants
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Age, Categorical
<=18 years
3 Participants
n=5 Participants
Age, Categorical
Between 18 and 65 years
13 Participants
n=5 Participants
Age, Categorical
>=65 years
0 Participants
n=5 Participants
Sex: Female, Male
Female
0 Participants
n=5 Participants
Sex: Female, Male
Male
16 Participants
n=5 Participants
Race/Ethnicity, Customized
White
16 Participants
n=5 Participants
Race/Ethnicity, Customized
Hispanic
1 Participants
n=5 Participants
Region of Enrollment
United States
16 Participants
n=5 Participants
Weight
149.3 lb
STANDARD_DEVIATION 192.9 • n=5 Participants

PRIMARY outcome

Timeframe: 5 days

BMD was measured as Z-scores/T-scores using Dual-Energy X-ray Absorptiometry (DEXA) scanning; specifically looking at Spine, Hip/Neck, and Hip total scores. BMD Z-scores compare what would be expected in someone your age and body size. A Z-score, is a unit of standard deviation, where above 0 would indicate the bones are more dense than expected, while a Z-score below 0 would indicate the bones were less dense.

Outcome measures

Outcome measures
Measure
Open Label
n=12 Participants
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Bone Biomarker Density (BMD)
Spine BMD (Spine L1-L4 Z-score)
-0.74 Z-Score
Standard Deviation 1.22
Bone Biomarker Density (BMD)
Hip Total BMD
-0.16 Z-Score
Standard Deviation 1.12
Bone Biomarker Density (BMD)
Hip Neck BMD
-0.16 Z-Score
Standard Deviation 1.23

PRIMARY outcome

Timeframe: 5 days

Hemophilia Joint Health Score (HJHS); with a higher score representing worst outcomes, scores could range from 0 (no problems) to a max score of 120 (severe problems).

Outcome measures

Outcome measures
Measure
Open Label
n=12 Participants
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Joint Health
19.91 score on a scale
Standard Deviation 16.26

PRIMARY outcome

Timeframe: 5 days

Visual Analog Scale (VAS) via the EQ-5D-3L was used to report participants self-rated health. EQ-5D-3L total score ranges from 5 (no problems) to 15 (significant problems). VAS scores could range from 0 (worst health ever) to 100 (best health ever).

Outcome measures

Outcome measures
Measure
Open Label
n=12 Participants
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Quality of Life Using the VAS and EQ-5D-3L
EQ-5D-3L
6.92 score on a scale
Standard Deviation 1.83
Quality of Life Using the VAS and EQ-5D-3L
VAS
82.42 score on a scale
Standard Deviation 15.08

PRIMARY outcome

Timeframe: 24 hours

Population: Some participant samples were unable to be analyzed at all time points due to poor sample quality or other factors.

cytokines were measured using ELISA/magnetic bead multiplex kits. We calculated concentration change from hour 0 to hour 24. Cytokines: FGF, C-Terminal telopeptide (CTX-1), Dickkopf WNT signaling pathway inhibitor 1(DKK1), Eotaxin, fibroblast growth factor 23(FGF23), interferon gamma, interleukin 13, interleukin 1 beta, interleukin receptor 1 antagonist, interleukin 2, interleukin 4, interleukin 6, interleukin 17, interleukin 8, interleukin 9 Insulin, interferon gamma induced protein 10 (IP10), Leptin, monocyte chemoattractant protein1, monocyte chemoattractant protein1, macrophage inflammatory protein 1a (MIP1a), osteoclasts, Osteoprotegerin, osteopontin, platelet derived growth factors, parathyroid horomone, Recepter activator of nuclear factor kappa-B ligand (RANKL), chemokine ligand 5, sclerostin, transforming growth factor-beta 1, transforming growth factor beta 2, transforming growth factor beta 3, tumor necrosis factor alpha, vascular endothelial growth factor, MIP1b.

Outcome measures

Outcome measures
Measure
Open Label
n=12 Participants
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
Basic FGF
2.04 pg/mL
Standard Deviation 1.78
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
C-Terminal telopeptide
0.03 pg/mL
Standard Deviation 0.12
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
DKK1
-7.73 pg/mL
Standard Deviation 15.63
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
Eotaxin
-2.84 pg/mL
Standard Deviation 14.09
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
fibroblast growth factor 23
-6.72 pg/mL
Standard Deviation 9.95
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
interferon gamma
0.12 pg/mL
Standard Deviation 0.52
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
interleukin 13
-0.76 pg/mL
Standard Deviation 1.52
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
Interleukin 1 beta
-0.12 pg/mL
Standard Deviation 0.19
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
interleukin receptor 1 antagonist
-131.72 pg/mL
Standard Deviation 429.93
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
interleukin 2
0.71 pg/mL
Standard Deviation 1.16
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
interleukin 4
-0.28 pg/mL
Standard Deviation 0.39
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
interleukin 6
0.22 pg/mL
Standard Deviation 0.38
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
interleukin 17
-0.13 pg/mL
Standard Deviation 0.80
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
interleukin 8
-0.56 pg/mL
Standard Deviation 0.99
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
interleukin 9
-14.83 pg/mL
Standard Deviation 14.03
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
Insulin
-279.39 pg/mL
Standard Deviation 1518.67
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
IP10
-10.06 pg/mL
Standard Deviation 36.49
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
Leptin
-208.39 pg/mL
Standard Deviation 6728.25
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
MCP1
-6.42 pg/mL
Standard Deviation 7.53
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
MIP1a
-0.23 pg/mL
Standard Deviation 0.40
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
osteoclasts
-2146.62 pg/mL
Standard Deviation 5921.06
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
osteoprotegerin
113.70 pg/mL
Standard Deviation 328.53
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
osteopontin
-493.87 pg/mL
Standard Deviation 6795.35
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
platelet derived growth factors
-103.83 pg/mL
Standard Deviation 156.52
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
parathyroid horomone
-6.72 pg/mL
Standard Deviation 25.70
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
RANKL
228.10 pg/mL
Standard Deviation 277.99
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
chemokine ligand 5
-414.77 pg/mL
Standard Deviation 681.82
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
sclerostin
-398.13 pg/mL
Standard Deviation 838.83
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
transforming growth factor-beta 1
-519.05 pg/mL
Standard Deviation 772.15
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
transforming growth factor beta - 2
-10.08 pg/mL
Standard Deviation 16.43
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
transforming growth factor beta - 3
-0.74 pg/mL
Standard Deviation 1.49
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
tumor necrosis factor alpha
-1.09 pg/mL
Standard Deviation 2.76
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
VEGF
-4.03 pg/mL
Standard Deviation 12.17
Plasma Cytokine Concentration Differences From 0-hour to 24-hour
MIP1b
-7.19 pg/mL
Standard Deviation 9.36

OTHER_PRE_SPECIFIED outcome

Timeframe: 5 days

VERITAS instrument self-reported compliance with factor replacement regimen. The VERITAS is composed of 6 subsections, where scores range from 1 (best adherence) to 5 (worst adherence). A total score was calculated with the minimum score of 24 (most adherent) and a maximum score of 120 (least adherent)

Outcome measures

Outcome measures
Measure
Open Label
n=12 Participants
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Medication Adherence
56.25 score on a scale
Standard Deviation 5.29

OTHER_PRE_SPECIFIED outcome

Timeframe: 5 days

The HAL is used to assess physical activity; scores range from 42 (severe problems) to 252 (no problems). The ISTH-BAT was used to evaluate bleeding phenotype. Each section was scored using a 0 to 4 scale for each of the 12 subsections. A total score was calculated by taking the sum of each section, resulting in a range of scores from 0 (no bleeding history) to 48 (most extensive clinical intervention for bleeding)

Outcome measures

Outcome measures
Measure
Open Label
n=12 Participants
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Hemophilia Activities List (HAL) and the International Society of Thrombosis and Hemostasis- Bleeding Assessment Tool (ISTH-BAT)
HAL
226 score on a scale
Standard Deviation 41.78
Hemophilia Activities List (HAL) and the International Society of Thrombosis and Hemostasis- Bleeding Assessment Tool (ISTH-BAT)
ISTH-BAT
16.92 score on a scale
Standard Deviation 6.56

OTHER_PRE_SPECIFIED outcome

Timeframe: 5 days

Haem-A-QoL was used to assess various components indicating participants quality of life. Haem-A-QoL is composed of 10 subsections with 3-8 questions in each. Questions are rated from 1 to 5, with 5 being the most negative influence on quality of life. Some questions are worded in the inverse, such that the 1 corresponds to the greatest negative impact on quality of life. In this case, the score is inverted to match the remainder of questions for statistical analysis. A Transformed scores is calculated for each subsection and for the total of all the subjections. The scores range from 0 (best quality of life) to 225 (worst quality of life).

Outcome measures

Outcome measures
Measure
Open Label
n=12 Participants
Everyone receives Advate (antihemophilic factor) on Day 1 and 3. Advate: Patients who are currently taking Advate as their factor replacement will be eligible for the 5-day study.
Participant Quality of Life
113.83 score on a scale
Standard Deviation 58.19

Adverse Events

Open Label

Serious events: 0 serious events
Other events: 0 other events
Deaths: 0 deaths

Serious adverse events

Adverse event data not reported

Other adverse events

Adverse event data not reported

Additional Information

Dr. Jayson Taylor

Oregon Health & Science University

Phone: 503-494-8716

Results disclosure agreements

  • Principal investigator is a sponsor employee
  • Publication restrictions are in place