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Safety, Pharmacokinetics and Pharmacodynamics of BPS804 in Osteogenesis Imperfecta

Phase 2
Completed
Conditions
Osteogenesis Imperfecta
Interventions
Registration Number
NCT01417091
Lead Sponsor
Ultragenyx Pharmaceutical Inc
Brief Summary

This is a randomized, open label intra-patient dose escalation study to evaluate safety and tolerability, pharmacokinetics, and pharmacodynamics of BPS804 in adults with osteogenesis imperfecta (OI).

Pharmacodynamic effect will be determined by serological biomarkers and radiologic assessments. In addition, tolerability and pharmacokinetics (PK) will be evaluated.

Detailed Description

This study was previously posted by Mereo BioPharma and was transferred to Ultragenyx in February 2021.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
10
Inclusion Criteria
  • Osteogenesis imperfecta
  • Two or more previous fractures
  • Bone mineral density Z-score of ≤ -1.0 and > -4.0
Exclusion Criteria
  • Open epiphyses
  • Fracture within last 2 weeks
  • Treatment with bisphosphonates/teriparatide (last 6 months)
  • Surgery within last year

Other protocol-defined inclusion/exclusion criteria may apply

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
Treatment groupBPS804-
Primary Outcome Measures
NameTimeMethod
Change in Z-score from baseline to Day 141Day 1 and Day 141

Bone mineral density will be assessed by dual-energy X-ray absorptiometry of the lumbar spine. Analysis will include four vertebral levels from L1 to L4. Individual vertebral levels may be excluded due to artifact. Bone mineral density Z-scores will be used as these are a comparison of a patient's BMD to that of a patient of the same age, sex, and ethnicity. The comparison of change from baseline with the matching change in the reference group will be done by 2-sample t-tests (1-sided). It will be considered a sign for efficacy, if any of the above show significant (2-sided, alpha=0.05) increase versus baseline in the BPS804 group on day 141.

Determination of pharmacodynamic effect by means of biomarkersDay 1 and Day 43

Biomarker data from serum bone formation biomarkers: procollagen type I N-terminal propeptide, procollagen type I C-terminal propeptide, osteocalcin, and bone-specific alkaline phosphatase, and from serum bone resporption mbiomarkers: C-telopeptides of type I collagen cross-links, and N-telopeptides of type I collagen cross-links will be reported as concentration results, measured using a specific assay with a working range defined by the Lower limit of quantification and Upper limit of quantification. It will be considered a sign for efficacy, if any of the above show significant (2-sided, alpha=0.05) increase versus baseline in the BPS804 group on day 43.

Safety and tolerability (composite outcome: standard laboratory, (serious) adverse events)Day 1 through 141

The assessment of safety will be based on primarily on the frequency of adverse events, laboratory abnormalities, and serious adverse events suspected by the investigators to be related to study treatments. The AE collection period extends from the time of first study drug administration drug until study completion. The intensity of each AE will be characterized and classified into 1 of the 3 generic categories (mild, moderate, or severe). The number and percentage of patients with adverse events will be tabulated by treatment group, body system and preferred term. The periods for adverse event tabulation will be from dose administration up to next dose administration if a further dose is given, and from dose administration to EOS for the last dose administration of a patient.

Secondary Outcome Measures
NameTimeMethod
Determination of the time of maximal concentration after the second doseDay 15 (prior to administration and 1x after administration) and Day 16

The time of maximal concentration after the second dose will be derived using using non-compartmental methods from data collected from days 15 (2x) and 16.

Determination of the area under the serum concentration-time curve from time zero to infinity after the third doseDays 29 (prior to administration and 3x after administration) and 30, 36, 43, 57, 85, 113, and 141

The area under the serum concentration-time curve from time zero to the end of the dosing interval tau after the third dose will be derived using non-compartmental methods from data collected on Days 29 (4x), 30, 36, 43, 57, 85, 113, and 141.

Determination of the maximal serum concentration after the third doseDays 29 (prior to administration and 3x after administration) and 30, 36, 43, 57, 85, 113, and 141

The maximal serum concentration after the third dose will be derived using using non-compartmental methods from data collected on Days 29 (4x), 30, 36, 43, 57, 85, 113, and 141.

Determination of the area under the serum concentration-time curve from time zero to the end of the dosing interval tau after the first doseDay 1 (prior to administration and 3x after administration) and Days 2, and 8

The area under the serum concentration-time curve from time zero to the end of the dosing interval tau after the first dose will be derived using using non-compartmental methods from data collected on Day 1 (4x) and Days 2, and 8.

Determination of the maximal concentration after the first doseDay 1 (prior to administration and 3x after administration) and Days 2, and 8

The maximal concentration after the first dose will be derived using using non-compartmental methods from data collected Day 1 (4x) and Days 2, and 8.

Determination of the maximal concentration after the second doseDay 15 (prior to administration and 1x after administration) and Day 16

The maximal concentration after the second dose will be derived using using non-compartmental methods from data collected from days 15 (2x) and 16.

Determination of the area under the serum concentration-time curve from time zero to the time of the last quantifiable concentration after the third doseDays 29 (prior to administration and 3x after administration) and 30, 36, 43, 57, 85, 113, and 141

The area under the serum concentration-time curve from time zero to the time of the last quantifiable concentration after the third dose will be derived using non-compartmental methods from data collected on Days 29 (4x), 30, 36, 43, 57, 85, 113, and 141.

Determination of the area under the serum concentration-time curve from time zero to the end of the dosing interval tau after the third doseDays 29 (prior to administration and 3x after administration) and 30, 36, 43, 57, 85, 113, and 141

The area under the serum concentration-time curve from time zero to the end of the dosing interval tau after the third dose will be derived using non-compartmental methods from data collected on Days 29 (4x), 30, 36, 43, 57, 85, 113, and 141.

Determination of the time of maximal concentration after the third doseDays 29 (prior to administration and 3x after administration) and 30, 36, 43, 57, 85, 113, and 141

The time of maximal concentration after the third dose will be derived using using non-compartmental methods from data collected on Days 29 (4x), 30, 36, 43, 57, 85, 113, and 141.

Determination of the serum concentration-time profiles of BPS804Day 1 (prior to administration and 3x after administration) and Days 2, 8, 15 (2x), 16, 29 (4x), 30, 36, 43, 57, 85, 113,141

Individual and overlaying individual serum concentration-time profiles of BPS804 will be constructed from the serial sampling on Day 1 (prior to administration and 3x after administration) and Days 2, 8, 15 (2x), 16, 29 (4x), 30, 36, 43, 57, 85, 113,141. In addition arithmetic and geometric mean serum concentration-time profiles of BPS804 will be constructed from the above data points.

Determination of the time of maximal concentration after the first doseDay 1 (prior to administration and 3x after administration) and Days 2, and 8

The time of maximal concentration after the first dose will be derived using using non-compartmental methods from data collected Day 1 (4x) and Days 2, and 8.

Determination of the terminal elimination half-life after the third doseDays 29 (prior to administration and 3x after administration) and 30, 36, 43, 57, 85, 113, and 141

The terminal elimination half-life after the third dose will be derived from data collected on Days 29 (4x), 30, 36, 43, 57, 85, 113, and 141 using a compound-specific modeling approach.

Determination of the concentration of total sclerostin in serumScreening, Days 1, 8, 15, 29, 36, 43, 57, 85, 113, 141

The function of sclerostin is described as an endogenous negative regulator of bone formation. Total serum sclerostin will be measured from samples collected at screening and on days 1, 8, 15, 29, 36, 43, 57, 85, 113, and 141

Immunogenicity evaluation in serumDays 1, 29, 85, and 141

Immunogenicity will only be assessed in patients randomized to the treatment group. Anti-BPS804 antibodies will be measured in human serum on Days 1, 29, 85, and 141 An immunogenicity positive patient at end of study will be followed up until anti-BPS804 antibody levels are back to levels measured on Day 1.

Trial Locations

Locations (1)

Novartis Investigative Site

🇩🇪

Wuerzburg, Germany

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