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NCT00446550

A Study of Oral AT2101 (Afegostat Tartrate) in Treatment-naive Patients With Gaucher Disease

Completed Phase 2 Results posted Last updated 15 August 2018
What this trial tests

Phase 2 trial testing afegostat tartrate in Gaucher Disease in 19 participants. Completed in 20 August 2009.

Timeline
11 June 2008
Primary endpoint
20 August 2009
20 August 2009

Quick facts

Lead sponsorAmicus Therapeutics
PhasePhase 2
StatusCompleted
Study typeINTERVENTIONAL
Allocationrandomized
Designparallel
Maskingnone
Primary purposetreatment
Enrollment19
Start date11 June 2008
Primary completion20 August 2009
Estimated completion20 August 2009
Sites11 locations across South Africa, United Kingdom, United States, Israel

Drugs / interventions tested

Conditions studied

Sponsor

Amicus Therapeutics — full company profile →

Who can join

Adults 18 to 74, any sex, with Gaucher Disease or Type 1 Gaucher Disease. Patients with the condition only — healthy volunteers not accepted.

Results — posted to ClinicalTrials.gov

Per-arm endpoint measurements with 95% confidence intervals where reported. Source: trial results section.

Number Of Participants Who Experienced Severe Treatment-emergent Adverse Events (TEAEs) Primary · Day 1 (after dosing) through Day 183

A TEAE was defined as any adverse event (AE) with start date on or after administration of study drug or pre-existing conditions that worsened on or after the start of the first study drug administration (on Day 1). A severe AE defined as an AE that was incapacitating and required medical intervention. The number of participants who experienced 1 or more severe TEAEs after dosing on Day 1 through the end of follow-up (Day 183) is presented. A summary of serious and all other non-serious AEs regardless of causality is located in the Reported Adverse Events module.

GroupValue95% CI
Afegostat Tartrate Treatment Regimen 12
Afegostat Tartrate Treatment Regimen 20
Change From Baseline To End Of Treatment In β-glucocerebrosidase (GCase) Levels In White Blood Cells (WBC) Secondary · Baseline, Day 169

GCase is a biomarker used to assess the PD effects of afegostat tartrate. Blood samples were collected to assess GCase levels in WBC. The baseline value was defined as the last non-missing value before the start of study drug.

GroupValue95% CI
Afegostat Tartrate Treatment Regimen 110.2± 15.87
Afegostat Tartrate Treatment Regimen 23.9± 4.12

Adverse events — posted to ClinicalTrials.gov

Time frame: Day 1 (after dosing) to Day 183. Reporting threshold: 5%. Adverse-event reports describe events observed during the trial — not all are caused by the drug.

Afegostat Tartrate Treatment Regimen 1
Serious: 0/11 (0%)
Deaths:
Afegostat Tartrate Treatment Regimen 2
Serious: 0/8 (0%)
Deaths:
Other adverse events (67 terms — click to expand)

ReactionSystemAfegostat Tartrate Treatme…Afegostat Tartrate Treatme…
Lacrimation increasedEye disorders
Dry eyeEye disorders
Eye irritationEye disorders
ArthralgiaMusculoskeletal and connective tissue disorders
SplenomegalyBlood and lymphatic system disorders
ThrombocytopeniaBlood and lymphatic system disorders
Abnormal sensation in eyeEye disorders
ChalazionEye disorders
Conjunctival haemorrhageEye disorders
Conjunctival irritationEye disorders
ConjunctivitisEye disorders
Eye inflammationEye disorders
Eye pruritusEye disorders
Vision blurredEye disorders
Abdominal distensionGastrointestinal disorders
Abdominal painGastrointestinal disorders
Anal fissureGastrointestinal disorders
ConstipationGastrointestinal disorders
DiarrhoeaGastrointestinal disorders
FlatulenceGastrointestinal disorders
Gastrointestinal disorderGastrointestinal disorders
Gingival bleedingGastrointestinal disorders
Oral painGastrointestinal disorders
Rectal haemorrhageGastrointestinal disorders
Tooth impactedGastrointestinal disorders
ToothacheGastrointestinal disorders
VomitingGastrointestinal disorders
Adverse drug reactionGeneral disorders
FatigueGeneral disorders
CholelithiasisHepatobiliary disorders
HepatomegalyHepatobiliary disorders
Eyelid infectionInfections and infestations
Fungal rashInfections and infestations
Gastroenteritis viralInfections and infestations
InfluenzaInfections and infestations
Upper respiratory tract infectionInfections and infestations
ContusionInjury, poisoning and procedural complications
Bacteria urineInvestigations
Vitamin B12 decreasedInvestigations
Intervertebral disc protrusionMusculoskeletal and connective tissue disorders

Data from ClinicalTrials.gov NCT00446550 adverse events section.

Sponsor's own description

This study evaluated the safety and tolerability of afegostat tartrate in participants with type 1 Gaucher disease who were not receiving enzyme replacement therapy (ERT) or substrate reduction therapy (SRT).

Publications & conference data

6 peer-reviewed publications reference this trial (live from Europe PMC):

  1. Therapeutics in the Pipeline Targeting <i>α</i>-Synuclein for Parkinson's Disease.
    Grosso Jasutkar H, Oh SE, Mouradian MM. · · 2022 · cited 69× · PMID 35017177 · DOI 10.1124/pharmrev.120.000133
  2. Therapeutic Approaches in Lysosomal Storage Diseases.
    Fernández-Pereira C, San Millán-Tejado B, Gallardo-Gómez M, Pérez-Márquez T, et al · · 2021 · cited 43× · PMID 34944420 · DOI 10.3390/biom11121775
  3. Lysosomal dysfunction in α-synuclein pathology: molecular mechanisms and therapeutic strategies.
    Dai L, Liu M, Ke W, Chen L, et al · · 2024 · cited 17× · PMID 39223418 · DOI 10.1007/s00018-024-05419-5
  4. Pharmacological Chaperones and Protein Conformational Diseases: Approaches of Computational Structural Biology.
    Grasso D, Galderisi S, Santucci A, Bernini A. · · 2023 · cited 16× · PMID 36982893 · DOI 10.3390/ijms24065819
  5. Precision Medicine in Parkinson's Disease: From Genetic Risk Signals to Personalized Therapy.
    Straccia G, Colucci F, Eleopra R, Cilia R. · · 2022 · cited 9× · PMID 36291241 · DOI 10.3390/brainsci12101308
  6. Lysosomal Dysfunction: Connecting the Dots in the Landscape of Human Diseases.
    Uribe-Carretero E, Rey V, Fuentes JM, Tamargo-Gómez I. · · 2024 · cited 8× · PMID 38248465 · DOI 10.3390/biology13010034

Verify or expand the search:

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Other Amicus Therapeutics trials

Trials by the same sponsor.

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Primary sources · FDA · ClinicalTrials.gov · EMA · SEC EDGAR · ChEMBL · Wikidata · full sourcing