Exploring triheptanoin as treatment for short chain enoyl CoA hydratase deficiency

Ann Clin Transl Neurol. 2021 May;8(5):1151-1157. doi: 10.1002/acn3.51359. Epub 2021 May 1.

Abstract

We explored the benefits of triheptanoin as a treatment for Short Chain Enoyl Co-A Hydratase (SCEH) deficiency. One child with early onset, severe SCEH Deficiency was treated with triheptanoin, an odd chain oil with anapleurotic properties, for 37 months. Blood and urine chemistry safety measures, motor skills assessment, physical exam, and neurological assessment were monitored over a 27 month period. Modest sustained gains in motor skills, attention, muscle bulk, and strength were observed without any significant adverse effects. Triheptanoin appears to be a promising effective treatment for SCEH Deficiency.

Publication types

  • Case Reports

MeSH terms

  • Brain Diseases, Metabolic, Inborn / drug therapy*
  • Brain Diseases, Metabolic, Inborn / metabolism
  • Brain Diseases, Metabolic, Inborn / physiopathology
  • Child
  • Enoyl-CoA Hydratase / deficiency*
  • Female
  • Humans
  • Triglycerides / administration & dosage
  • Triglycerides / pharmacology*

Substances

  • Triglycerides
  • triheptanoin
  • Enoyl-CoA Hydratase