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A Study to Assess the Safety and Tolerability of Lucerastat in Subjects With Fabry Disease

Phase 1
Completed
Conditions
Fabry Disease
Interventions
Drug: Enzyme replacement therapy (ERT)
Registration Number
NCT02930655
Lead Sponsor
Idorsia Pharmaceuticals Ltd.
Brief Summary

The primary purpose of this study was to assess the safety and tolerability of lucerastat in adults with Fabry Disease receiving Enzyme Replacement Therapy (ERT).

The secondary objectives were to investigate the effects of lucerastat on plasma and urine levels of biomarkers, to assess its effects on renal and cardiac functions and to determine the pharmacokinetic profile of lucerastat at steady-state.

Detailed Description

Not available

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
14
Inclusion Criteria
  • Signed informed consent form
  • Male and female adult subjects with a diagnosis of Fabry Disease (FD) based on historical assessments (residual α-GAL A activity level below lower limit of normal for males and presence of a galactosidase alpha mutation for females) and a history of clinical symptoms of FD
  • On ERT for at least 24 months without any change in dose within the last 6 months prior to screening
Exclusion Criteria
  • Severe renal function impairment
  • Severe residual neurologic deficit
  • Clinically significant unstable cardiac disease
  • Any circumstances or conditions, which, in the opinion of the investigator, may have affected full participation in the study or compliance with the protocol

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
Lucerastat groupLucerastatTen subjects with Fabry Disease received 1000 mg of oral lucerastat twice daily for 12 weeks in addition to their standard of care treatment (enzyme replace therapy).
Lucerastat groupEnzyme replacement therapy (ERT)Ten subjects with Fabry Disease received 1000 mg of oral lucerastat twice daily for 12 weeks in addition to their standard of care treatment (enzyme replace therapy).
Control groupEnzyme replacement therapy (ERT)Four subjects with Fabry Disease under enzyme replace therapy (ERT) as standard of care treatment were included as a control group.
Primary Outcome Measures
NameTimeMethod
Change from baseline in blood pressureUp to Week 12
Change from baseline in heart rateUp to Week 12
Change from baseline in electrocardiogram (ECG) variablesUp to Week 12

The duration (in ms) of the different ECG variables were measured using a standard 12-lead ECG

Change from baseline in body weightUp to Week 12
Number of subjects with treatment-emergent adverse events and serious adverse eventsUp to Week 12
Number of subjects with adverse events leading to premature discontinuation of lucerastat or ERTUp to Week 12
Number of subjects with treatment-emergent abnormalities in laboratory variablesUp to Week 12
Secondary Outcome Measures
NameTimeMethod
Change from baseline in urine albumin-to-creatinine ratio (UACR)Up to Week 12

UACR was used to monitor renal function in subjects with Fabry Disease

Terminal half-life [t(1/2)]of lucerastatAt Week 4 visit, blood samples drawn at the following time points: pre-dose, 0.5h, 1h, 1.5h, 2h, 2.5h, 3h, 3.5h, 4h, 6h, 8h, 10h, 12h post-dose
Change from baseline in left ventricular ejection fraction (LVEF)Up to Week 12

LVEF was used to monitor cardiac function in subjects with Fabry Disease

Change from baseline in estimated glomerular filtration rate (eGFR)Up to Week 12

eGFR was used to monitor renal function in subjects with Fabry Disease

Change from baseline in plasma biomarkers of Fabry DiseaseUp to Week 12

Biomarkers reflecting glycolipid metabolism were measured (unit of measure: ng/mL)

Time to reach Cmax (tmax) of lucerastatAt Week 4 visit, blood samples drawn at the following time points: pre-dose, 0.5h, 1h, 1.5h, 2h, 2.5h, 3h, 3.5h, 4h, 6h, 8h, 10h, 12h post-dose

tmax was determined directly from the observed plasma concentration-time curves of lucerastat. Blood samples for PK analyses were drawn at scheduled time points at the Week 4 visit

Change from baseline in urine biomarker of Fabry DiseaseUp to Week 12

Biomarker reflecting glycolipid metabolism was measured (unit of measure: ng/mg)

Change from baseline in left ventricular mass index (LVMi)Up to Week 12

LVMi was used to monitor cardiac function in subjects with Fabry Disease

Maximum plasma concentration (Cmax) of lucerastatAt Week 4 visit, blood samples drawn at the following time points: pre-dose, 0.5h, 1h, 1.5h, 2h, 2.5h, 3h, 3.5h, 4h, 6h, 8h, 10h, 12h post-dose

Cmax was determined directly from the observed plasma concentration-time curves of lucerastat. Blood samples for PK analyses were drawn at scheduled time points at the Week 4 visit

Area under the plasma concentration-time curve [AUC(tau)] of lucerastatAt Week 4 visit, blood samples drawn at the following time points: pre-dose, 0.5h, 1h, 1.5h, 2h, 2.5h, 3h, 3.5h, 4h, 6h, 8h, 10h, 12h post-dose

AUC(tau) corresponds to the area under the plasma concentration time curve of lucerastat over a dosing interval (tau = 12 hours)

Trial Locations

Locations (1)

Investigator Site

🇩🇪

Würzburg, Germany

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