Status:

COMPLETED

A Study of Intrathecal Enzyme Therapy for Cognitive Decline in MPS I

Lead Sponsor:

Patricia I. Dickson, M.D.

Collaborating Sponsors:

The Ryan Foundation

BioMarin Pharmaceutical

Conditions:

Cognitive Decline

Mucopolysaccharidosis I

Eligibility:

All Genders

6+ years

Phase:

NA

Brief Summary

This is a 24-month study of the use of laronidase administered into the spinal fluid to treat cognitive decline in mucopolysaccharidosis I (MPS I). MPS I is a rare genetic condition due to deficiency ...

Detailed Description

This study is a 24-month open label, prospective, randomized trial in 16 MPS I patients age six years or older who have documented evidence of cognitive decline. The study will test the safety and eff...

Eligibility Criteria

Inclusion

  • The presence of MPS I disease as documented by low α-L-iduronidase activity
  • Age six years or older.
  • The presence of acquired cognitive deficits as demonstrated by:
  • A score of one standard deviation below mean on IQ testing or in one domain of neuropsychological function (language, memory, or non-verbal ability), OR
  • Documented historical evidence of a decline of greater than one standard deviation on sequential testing, OR
  • A score between 0.75 and 1 standard deviation below the mean, AND the cognitive deficit affects daily performance.
  • The decline in function is not explainable by other neurological or psychiatric factors.
  • Subject and/or guardian willing and able to provide written informed consent.
  • Negative urine pregnancy test at screening (non-sterile females of child-bearing potential only)
  • Currently using two acceptable methods of birth control as determined by the investigator and willing to continue to use acceptable birth control during their participation in the study (non-sterile females of child-bearing potential who are sexually active only)
  • Willing and able to comply with study procedures. For example, the subjects must be able to complete written and computer-based testing. The subjects must be able to lie still in the MRI scanner for at least 40 minutes without sedation.

Exclusion

  • The subject has undergone hematopoietic stem cell transplantation
  • Recent initiation of intravenous Aldurazyme® therapy with less than 6 months of therapy. Subjects who have been receiving Aldurazyme® therapy for more than 6 months, and those who have never received Aldurazyme® therapy, will be allowed to enroll
  • Pregnant or lactating, or considering pregnancy
  • Receipt of an investigational drug or procedure within 30 days of enrollment
  • A condition, medical or other, that prevents participation in the study, including severe auditory or visual impairment, significant lumbar pathology, lumbar catheter, or recent major surgery within 6 weeks that would preclude their ability to participate.
  • Infusion reactions to intravenous Aldurazyme® therapy that require ongoing medical intervention, special prophylaxis or altered rate or dose of enzyme administration
  • The subject has a programmable VP shunt that is incompatible with the 3 Tesla MRI magnet and is unable or unwilling to undergo shunt revision to a MRI compatible device.
  • The subject has another contraindication for MRI, such as nonremovable metal in the body.
  • The subject has severely impaired spinal CSF flow, demonstrated by failure of appearance of 99mTechnetium-DTPA in the basal cisterns by 4 hours after intra-lumbar administration.

Key Trial Info

Start Date :

June 1 2009

Trial Type :

INTERVENTIONAL

Allocation :

ACTUAL

End Date :

April 1 2015

Estimated Enrollment :

9 Patients enrolled

Trial Details

Trial ID

NCT00852358

Start Date

June 1 2009

End Date

April 1 2015

Last Update

January 26 2016

Active Locations (3)

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Page 1 of 1 (3 locations)

1

Children's Hospital & Research Center Oakland

Oakland, California, United States, 94609-1809

2

Los Angeles Biomedical Institute at Harbor-UCLA

Torrance, California, United States, 90502

3

University of Minnesota

Minneapolis, Minnesota, United States, 55455