Study of the Effects of Oral AT1001 (Migalastat Hydrochloride) in Patients With Fabry Disease

NCT ID: NCT00925301

Last Updated: 2018-10-30

Study Results

Results available

Outcome measurements, participant flow, baseline characteristics, and adverse events have been published for this study.

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Basic Information

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Recruitment Status

COMPLETED

Clinical Phase

PHASE3

Total Enrollment

67 participants

Study Classification

INTERVENTIONAL

Study Start Date

2009-10-23

Study Completion Date

2014-01-29

Brief Summary

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The primary objective of this study was to compare the effect of migalastat (123 milligrams \[mg\] of migalastat \[equivalent to 150 mg of migalastat hydrochloride\]) (migalastat) versus placebo on kidney globotriaosylceramide (GL-3).

Detailed Description

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This double-blind, randomized, placebo-controlled study was conducted in 67 participants at 46 sites worldwide. The study consisted of 2 stages and an optional open-label treatment extension phase:

Stage 1 included a screening period of up to 2 months followed by a 6-month treatment period which involved 4 visits to the clinic. Participants were randomized in equal proportions to receive either migalastat or placebo.

After completing the 6-month double-blind phase, all participants entered Stage 2 of the study and received migalastat in an open-label manner. Stage 2 treatment lasted for 6 months and involved up to 4 visits to the clinic.

Participants who completed both Stage 1 and Stage 2 of the study as scheduled were offered the opportunity to participate in an open-label treatment extension phase with migalastat. The open-label treatment extension phase lasted 12 months and involved 2 visits to the clinic. A follow-up visit was undertaken 1 month following completion or discontinuation from the open-label treatment extension. Participants completing the 12-month open-label treatment extension and providing consent to enter a separate long-term extension were not required to complete this follow-up visit.

Study assessments included clinical laboratory tests, 12-lead electrocardiogram, kidney biopsy, kidney function testing, echocardiography, and patient-reported outcomes.

Conditions

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Fabry Disease

Study Design

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Allocation Method

RANDOMIZED

Intervention Model

PARALLEL

Primary Study Purpose

TREATMENT

Blinding Strategy

DOUBLE

Participants Investigators
Sponsor and Assessor were also blinded

Study Groups

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Migalastat

Migalastat 150-mg capsule taken orally every other day (QOD) for 6 months and an open-label 6-month treatment extension, followed by an optional, 12-month, open-label treatment extension.

Group Type EXPERIMENTAL

migalastat hydrochloride

Intervention Type DRUG

Oral capsule QOD

Placebo

Placebo capsule taken orally QOD for 6 months.

Group Type PLACEBO_COMPARATOR

Placebo

Intervention Type DRUG

Oral capsule QOD

Interventions

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migalastat hydrochloride

Oral capsule QOD

Intervention Type DRUG

Placebo

Oral capsule QOD

Intervention Type DRUG

Other Intervention Names

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AT1001 Galafold Migalastat

Eligibility Criteria

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Inclusion Criteria

* Male or female between the ages of 16 and 74 diagnosed with Fabry disease.
* Confirmed mutant form of α-galactosidase A shown to be responsive to migalastat in vitro.
* Participant has never been treated with enzyme replacement therapy (ERT) or has not received ERT for 6 consecutive months or longer before the screening visit for the study.
* Urine GL-3 ≥4 times the upper limit of normal at screening.
* Participants taking angiotensin converting enzyme inhibitors or angiotensin receptor blockers must be on a stable dose for a minimum of 4 weeks before the baseline visit.
* Females who can become pregnant and all males agree to be sexually abstinent or use medically accepted methods of birth control during the study and for 30 days after study completion.
* Participant is willing and able to provide written informed consent and assent, if applicable.

Exclusion Criteria

* Participant has undergone or is scheduled to undergo kidney transplantation, or is currently on dialysis.
* Estimated glomerular filtration rate \<30 milliliters per minute per 1.73 meters squared (chronic kidney disease Stage 4 or 5) based on the Modification of Diet in Renal Disease equation at screening.
* Pregnant or breast-feeding.
* History of allergy or sensitivity to study medication (including excipients) or other iminosugars (for example, miglustat, miglitol).
* Participant is treated or has been treated with any investigational drug within 30 days of study start.
* Participant is currently treated or has ever been treated with migalastat.
Minimum Eligible Age

16 Years

Maximum Eligible Age

74 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Amicus Therapeutics

INDUSTRY

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Principal Investigators

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Medical Monitor, Clinical Research

Role: STUDY_DIRECTOR

Amicus Therapeutics

Locations

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Los Angeles, California, United States

Site Status

San Francisco, California, United States

Site Status

Decatur, Georgia, United States

Site Status

Chicago, Illinois, United States

Site Status

Kansas City, Kansas, United States

Site Status

Boston, Massachusetts, United States

Site Status

Grand Rapids, Michigan, United States

Site Status

New York, New York, United States

Site Status

Pittsburgh, Pennsylvania, United States

Site Status

Dallas, Texas, United States

Site Status

Salt Lake City, Utah, United States

Site Status

Springfield, Virginia, United States

Site Status

Seattle, Washington, United States

Site Status

Buenos Aires, , Argentina

Site Status

Adelaide, , Australia

Site Status

Parkville, , Australia

Site Status

Porto Alegre, , Brazil

Site Status

São Paulo, , Brazil

Site Status

Montreal, Quebec, Canada

Site Status

Copenhagen, , Denmark

Site Status

Cairo, , Egypt

Site Status

Garches, , France

Site Status

Roma, , Italy

Site Status

Warsaw, , Poland

Site Status

Barcelona, , Spain

Site Status

Zaragoza, , Spain

Site Status

Ankara, , Turkey (Türkiye)

Site Status

Salford, , United Kingdom

Site Status

Countries

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United States Argentina Australia Brazil Canada Denmark Egypt France Italy Poland Spain Turkey (Türkiye) United Kingdom

References

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Germain DP, Nicholls K, Giugliani R, Bichet DG, Hughes DA, Barisoni LM, Colvin RB, Jennette JC, Skuban N, Castelli JP, Benjamin E, Barth JA, Viereck C. Efficacy of the pharmacologic chaperone migalastat in a subset of male patients with the classic phenotype of Fabry disease and migalastat-amenable variants: data from the phase 3 randomized, multicenter, double-blind clinical trial and extension study. Genet Med. 2019 Sep;21(9):1987-1997. doi: 10.1038/s41436-019-0451-z. Epub 2019 Feb 6.

Reference Type DERIVED
PMID: 30723321 (View on PubMed)

Schiffmann R, Bichet DG, Jovanovic A, Hughes DA, Giugliani R, Feldt-Rasmussen U, Shankar SP, Barisoni L, Colvin RB, Jennette JC, Holdbrook F, Mulberg A, Castelli JP, Skuban N, Barth JA, Nicholls K. Migalastat improves diarrhea in patients with Fabry disease: clinical-biomarker correlations from the phase 3 FACETS trial. Orphanet J Rare Dis. 2018 Apr 27;13(1):68. doi: 10.1186/s13023-018-0813-7.

Reference Type DERIVED
PMID: 29703262 (View on PubMed)

Benjamin ER, Della Valle MC, Wu X, Katz E, Pruthi F, Bond S, Bronfin B, Williams H, Yu J, Bichet DG, Germain DP, Giugliani R, Hughes D, Schiffmann R, Wilcox WR, Desnick RJ, Kirk J, Barth J, Barlow C, Valenzano KJ, Castelli J, Lockhart DJ. The validation of pharmacogenetics for the identification of Fabry patients to be treated with migalastat. Genet Med. 2017 Apr;19(4):430-438. doi: 10.1038/gim.2016.122. Epub 2016 Sep 22.

Reference Type DERIVED
PMID: 27657681 (View on PubMed)

Germain DP, Hughes DA, Nicholls K, Bichet DG, Giugliani R, Wilcox WR, Feliciani C, Shankar SP, Ezgu F, Amartino H, Bratkovic D, Feldt-Rasmussen U, Nedd K, Sharaf El Din U, Lourenco CM, Banikazemi M, Charrow J, Dasouki M, Finegold D, Giraldo P, Goker-Alpan O, Longo N, Scott CR, Torra R, Tuffaha A, Jovanovic A, Waldek S, Packman S, Ludington E, Viereck C, Kirk J, Yu J, Benjamin ER, Johnson F, Lockhart DJ, Skuban N, Castelli J, Barth J, Barlow C, Schiffmann R. Treatment of Fabry's Disease with the Pharmacologic Chaperone Migalastat. N Engl J Med. 2016 Aug 11;375(6):545-55. doi: 10.1056/NEJMoa1510198.

Reference Type DERIVED
PMID: 27509102 (View on PubMed)

Bichet DG, Aerts JM, Auray-Blais C, Maruyama H, Mehta AB, Skuban N, Krusinska E, Schiffmann R. Assessment of plasma lyso-Gb3 for clinical monitoring of treatment response in migalastat-treated patients with Fabry disease. Genet Med. 2021 Jan;23(1):192-201. doi: 10.1038/s41436-020-00968-z. Epub 2020 Sep 30.

Reference Type DERIVED
PMID: 32994552 (View on PubMed)

Other Identifiers

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FACETS

Identifier Type: OTHER

Identifier Source: secondary_id

2009-013459-31

Identifier Type: EUDRACT_NUMBER

Identifier Source: secondary_id

AT1001-011

Identifier Type: -

Identifier Source: org_study_id

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