A One Visit Follow Up of Adults With Fabry Disease Who Started Long-term Enzyme Replacement Therapy As Children

NCT ID: NCT04002531

Last Updated: 2019-11-22

Study Results

Results pending

The study team has not published outcome measurements, participant flow, or safety data for this trial yet. Check back later for updates.

Basic Information

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Recruitment Status

UNKNOWN

Clinical Phase

NA

Total Enrollment

12 participants

Study Classification

INTERVENTIONAL

Study Start Date

2018-11-10

Study Completion Date

2019-12-31

Brief Summary

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The objective of this study is to obtain follow up data on a cohort of well-studied patients with Fabry disease who have been on ERT since childhood for a total of about 15 years.

Detailed Description

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The long-term effect of initiating ERT in childhood is unknown. Prospective studies of Children with Fabry disease on 0.2 mg/kg agalsidase alfa every other week were performed. The patients were 7-17 years of age at initial study enrollment. The first open-label protocol was TKT023, a 6 months study (August 12, 2002-October 20, 2004) that was followed by an extension study TKT029 (June 10, 2004-June 15, 2011; ClinicalTrials.gov identifier NCT00084084). Since completing TKT029, all US patients were switched to commercial agalsidase beta. Therefore, these patients have now been treated for about 15 years.This study involves a one-visit follow up on these patients using the same protocol-driven studies as were used in TKT029. The long-term follow up data gathered will consist of a rare description of the disease profile in patients who were treated with ERT since childhood.

Conditions

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Quality of Life Renal Insufficiency Cardiac Event

Study Design

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Allocation Method

NA

Intervention Model

SINGLE_GROUP

Specific group of adults with Fabry disease who received Replagal infusions as children
Primary Study Purpose

OTHER

Blinding Strategy

NONE

Study Groups

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Single Visit

1. General and neurological examination
2. Vital signs including height, weight, blood pressure, pulse, temperature
3. 12 lead ECG
4. 2 hour Holter monitor for heart rate variability
5. Echocardiogram
6. Renal function will be assessed by the eGFR. The eGFR will be calculated from serum creatinine using CKD-EPI equation.
7. CBC with differential
8. Complete metabolic panel
9. Urinalysis
10. Urine Albumin/creatinine ratio.
11. Urine and plasma samples for biomarkers (Gb3, lyso-Gb3) that will be stored in -80 freezer and assayed in our lab.
12. Brief Pain Inventory questionnaire.
13. Quality of Life Questionnaires (SF36)

Group Type OTHER

General and Neurological examination

Intervention Type OTHER

Information about your general health, neurological symptoms and current medications with be collected

Vital signs

Intervention Type OTHER

Height, weight, blood pressure, heart rate, and respiratory rate and temperature will be measured.

12 lead electrocardiogram

Intervention Type PROCEDURE

A non-invasive test that measures the electrical activity of the heart

Echocardiogram

Intervention Type PROCEDURE

A non-invasive sonogram of the heart

Blood draw

Intervention Type PROCEDURE

Blood will be drawn to evaluate general health and renal function (kidney health)

Urine collection

Intervention Type PROCEDURE

Urine will be collection to evaluate renal function (kidney health)

2-hour Holter Monitor

Intervention Type PROCEDURE

A non-invasive test that measures the electrical activity of the heart continuously over 2 hours

Brief Pain Inventory questionnaire

Intervention Type OTHER

A questionnaire about daily pain

Quality of Life questionnaire

Intervention Type OTHER

A questionnaire about the impact of disease on their activities of daily living and quality of life

Interventions

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General and Neurological examination

Information about your general health, neurological symptoms and current medications with be collected

Intervention Type OTHER

Vital signs

Height, weight, blood pressure, heart rate, and respiratory rate and temperature will be measured.

Intervention Type OTHER

12 lead electrocardiogram

A non-invasive test that measures the electrical activity of the heart

Intervention Type PROCEDURE

Echocardiogram

A non-invasive sonogram of the heart

Intervention Type PROCEDURE

Blood draw

Blood will be drawn to evaluate general health and renal function (kidney health)

Intervention Type PROCEDURE

Urine collection

Urine will be collection to evaluate renal function (kidney health)

Intervention Type PROCEDURE

2-hour Holter Monitor

A non-invasive test that measures the electrical activity of the heart continuously over 2 hours

Intervention Type PROCEDURE

Brief Pain Inventory questionnaire

A questionnaire about daily pain

Intervention Type OTHER

Quality of Life questionnaire

A questionnaire about the impact of disease on their activities of daily living and quality of life

Intervention Type OTHER

Other Intervention Names

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MD assessment Blood pressure, heart rate, respiratory rate EKG, ECG Cardiac echo Blood collection, phlebotomy, lab test Holter BPI, Pain questionnaire SF 36

Eligibility Criteria

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Inclusion Criteria

1. Patients who participated in TKT029 and who are willing and able to come to Dallas for 1 visit for standard of care testing.
2. Sign the protocol informed consent form
3. Have been on continuous commercial ERT since TKT029 has ended

Exclusion Criteria

1. Patients who are unable to understand the nature, scope, and possible consequences of the study.
2. Patient does not give his written informed consent to participate in this study
3. Patient is unable to comply with the protocol, e.g., uncooperative with protocol schedule, refusal to agree to all of the study procedures.
4. Patient has been off ERT for an extended period of time as assessed by the investigator.
Minimum Eligible Age

18 Years

Eligible Sex

ALL

Accepts Healthy Volunteers

No

Sponsors

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Shire

INDUSTRY

Sponsor Role collaborator

Baylor Research Institute

OTHER

Sponsor Role lead

Responsible Party

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Responsibility Role SPONSOR

Locations

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Baylor University Medical Center

Dallas, Texas, United States

Site Status

Countries

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United States

References

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Schiffmann R. Fabry disease. Pharmacol Ther. 2009 Apr;122(1):65-77. doi: 10.1016/j.pharmthera.2009.01.003. Epub 2009 Feb 8.

Reference Type BACKGROUND
PMID: 19318041 (View on PubMed)

Schiffmann R, Ries M. Fabry Disease: A Disorder of Childhood Onset. Pediatr Neurol. 2016 Nov;64:10-20. doi: 10.1016/j.pediatrneurol.2016.07.001. Epub 2016 Jul 29.

Reference Type BACKGROUND
PMID: 27555236 (View on PubMed)

Echevarria L, Benistan K, Toussaint A, Dubourg O, Hagege AA, Eladari D, Jabbour F, Beldjord C, De Mazancourt P, Germain DP. X-chromosome inactivation in female patients with Fabry disease. Clin Genet. 2016 Jan;89(1):44-54. doi: 10.1111/cge.12613. Epub 2015 Jun 22.

Reference Type BACKGROUND
PMID: 25974833 (View on PubMed)

Dobyns WB. The pattern of inheritance of X-linked traits is not dominant or recessive, just X-linked. Acta Paediatr Suppl. 2006 Apr;95(451):11-5. doi: 10.1111/j.1651-2227.2006.tb02383.x.

Reference Type BACKGROUND
PMID: 16720459 (View on PubMed)

MacDermot KD, Holmes A, Miners AH. Natural history of Fabry disease in affected males and obligate carrier females. J Inherit Metab Dis. 2001;24 Suppl 2:13-4; discussion 11-2. doi: 10.1023/a:1012447102358. No abstract available.

Reference Type BACKGROUND
PMID: 11758673 (View on PubMed)

Schiffmann R, Warnock DG, Banikazemi M, Bultas J, Linthorst GE, Packman S, Sorensen SA, Wilcox WR, Desnick RJ. Fabry disease: progression of nephropathy, and prevalence of cardiac and cerebrovascular events before enzyme replacement therapy. Nephrol Dial Transplant. 2009 Jul;24(7):2102-11. doi: 10.1093/ndt/gfp031. Epub 2009 Feb 13.

Reference Type BACKGROUND
PMID: 19218538 (View on PubMed)

Kwon JM, Matern D, Kurtzberg J, Wrabetz L, Gelb MH, Wenger DA, Ficicioglu C, Waldman AT, Burton BK, Hopkins PV, Orsini JJ. Consensus guidelines for newborn screening, diagnosis and treatment of infantile Krabbe disease. Orphanet J Rare Dis. 2018 Feb 1;13(1):30. doi: 10.1186/s13023-018-0766-x.

Reference Type BACKGROUND
PMID: 29391017 (View on PubMed)

Meikle PJ, Hopwood JJ, Clague AE, Carey WF. Prevalence of lysosomal storage disorders. JAMA. 1999 Jan 20;281(3):249-54. doi: 10.1001/jama.281.3.249.

Reference Type BACKGROUND
PMID: 9918480 (View on PubMed)

Schiffmann R, Hughes DA, Linthorst GE, Ortiz A, Svarstad E, Warnock DG, West ML, Wanner C; Conference Participants. Screening, diagnosis, and management of patients with Fabry disease: conclusions from a "Kidney Disease: Improving Global Outcomes" (KDIGO) Controversies Conference. Kidney Int. 2017 Feb;91(2):284-293. doi: 10.1016/j.kint.2016.10.004. Epub 2016 Dec 18.

Reference Type BACKGROUND
PMID: 27998644 (View on PubMed)

Schiffmann R, Pastores GM, Lien YH, Castaneda V, Chang P, Martin R, Wijatyk A. Agalsidase alfa in pediatric patients with Fabry disease: a 6.5-year open-label follow-up study. Orphanet J Rare Dis. 2014 Nov 26;9:169. doi: 10.1186/s13023-014-0169-6.

Reference Type BACKGROUND
PMID: 25425121 (View on PubMed)

Related Links

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Other Identifiers

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018-706

Identifier Type: -

Identifier Source: org_study_id

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