Docosahexaenoic acid therapy in peroxisomal diseases: results of a double-blind, randomized trial

Neurology. 2010 Aug 31;75(9):826-30. doi: 10.1212/WNL.0b013e3181f07061.

Abstract

Objectives: Peroxisome assembly disorders are genetic disorders characterized by biochemical abnormalities, including low docosahexaenoic acid (DHA). The objective was to assess whether treatment with DHA supplementation would improve biochemical abnormalities, visual function, and growth in affected individuals.

Methods: This was a randomized, double-blind, placebo-controlled trial conducted at a single center. Treatment groups received supplements of DHA (100 mg/kg per day). The primary outcome measures were the change from baseline in the visual function and physical growth during the 1 year follow-up period.

Results: Fifty individuals were enrolled and randomized. Two were subsequently excluded from study analysis when it was determined that they had a single enzyme disorder of peroxisomal beta oxidation. Thirty-four returned for follow-up. Nine patients died during the trial of their disorder, and 5 others were lost to follow-up. DHA supplementation was well tolerated. There was no difference in the outcomes between the treated and untreated groups in biochemical function, electroretinogram, or growth. Improvements were seen in both groups in certain individuals.

Conclusions: DHA supplementation did not improve the visual function or growth of treated individuals with peroxisome assembly disorders.

Classification of evidence: This interventional study provides Class II evidence that DHA supplementation did not improve the visual function or growth of treated individuals with peroxisome assembly disorders during an average of 1 year of follow-up in patients aged 1 to 144 months.

Publication types

  • Comparative Study
  • Randomized Controlled Trial
  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Body Height / drug effects
  • Body Height / physiology
  • Child, Preschool
  • Dietary Supplements
  • Docosahexaenoic Acids / pharmacology
  • Docosahexaenoic Acids / therapeutic use*
  • Double-Blind Method
  • Electroretinography / drug effects
  • Follow-Up Studies
  • Humans
  • Infant
  • Infant, Newborn
  • Peroxisomal Disorders / drug therapy*
  • Peroxisomal Disorders / physiopathology
  • Refsum Disease, Infantile / drug therapy*
  • Refsum Disease, Infantile / physiopathology
  • Treatment Outcome
  • Visual Perception / drug effects
  • Visual Perception / physiology
  • Zellweger Syndrome / drug therapy*
  • Zellweger Syndrome / physiopathology

Substances

  • Docosahexaenoic Acids