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Phase 2 N=12 Treatment

Interferon Gamma-1b in Friedreich Ataxia (FRDA)

Friedreich Ataxia

Enrolled (actual)
12
Serious AEs
0.0%
Results posted
Apr 2015
Primary outcome: Primary: Change in Whole Blood Frataxin Levels — -1.5 percentage of baseline frataxin level — p=0.027

Study Design & Population

Study type
Interventional
Phase
Phase 2
Interventions
Interferon Gamma-1b (Drug)
Age
Pediatric · 5+ yrs
Sex
All
Sponsor
Children's Hospital of Philadelphia
Primary completion
Mar 2014

Outcome Measures

OutcomeResultp-value
PRIMARY
Change in Whole Blood Frataxin Levels
-1.5 0.027 sig
SECONDARY
Change in Total Friedreich Ataxia Rating Scale (FARS) Score
-4.98 0.0078 sig

Summary

Friedreich ataxia (FRDA) is a progressive neurodegenerative disease of children and adults for which there is presently no therapy. Recently, a study reported that interferon gamma (IFN-g) could raise frataxin protein levels in both cell lines derived from patients with Friedreich ataxia and in a mouse model with Friedreich ataxia. The present study will test whether IFN-g is safe, tolerated and potentially efficacious in a heterogeneous cohort of children with FRDA.

Eligibility Criteria

Inclusion Criteria

  • Subjects with FRDA confirmed by genetic testing with 2 expanded Guanine-adenine-adenine repeats
  • Females who are not pregnant or breast feeding, and who do not intend to become pregnant. Females of child-bearing potential must use a reliable method of contraception and must provide a negative urine pregnancy test at screening
  • Stable doses of all medications, vitamins and supplements for 30 days prior to study entry and for the duration of the study
  • Parent/guardian permission (informed consent) and child assent

Exclusion Criteria

  • Any unstable illness that in the investigator's opinion precludes participation in this study
  • Use of any investigational product within 30 days prior to enrollment
  • Subjects with a history of substance abuse
  • Presence of clinically significant cardiac disease
  • History of hypersensitivity to IFN-g or E. coli derived products
  • Presence of severe renal disease or hepatic disease
  • Clinically significant abnormal White blood cell count, hemoglobin or platelet count
  • Any subject planning a scheduled surgical procedure during the study
View full record on ClinicalTrials.gov →

Data sourced from ClinicalTrials.gov (NCT01965327). Outcome figures and adverse-event rates are extracted automatically from the registry's posted results and are provided for clinician reference, not as a substitute for the primary publication.

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