Mitochondrial neurogastrointestinal encephalomyopathy: novel pathogenic mutations in thymidine phosphorylase gene in two Italian brothers

Neuropediatrics. 2012 Aug;43(4):201-8. doi: 10.1055/s-0032-1315431. Epub 2012 May 22.

Abstract

Mitochondrial neurogastrointestinal encephalomyopathy (MNGIE, MIM 603041) is an autosomal recessive multisystem disorder occurring due to mutations in a nuclear gene coding for the enzyme thymidine phosphorylase (TYMP). Clinical features of MNGIE include gastrointestinal dysmotility, cachexia, ptosis or ophthalmoparesis, peripheral neuropathy, diffuse leukoencephalopathy, and signs of mitochondrial dysfunction in tissues. We report the clinical and molecular findings in two brothers in whom novel TYMP gene mutations (c.215-13_215delinsGCGTGA; c.1159 + 2T > A) were associated with different clinical presentations and outcomes.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Brain / pathology
  • DNA Mutational Analysis
  • Humans
  • Intestinal Pseudo-Obstruction / genetics*
  • Intestinal Pseudo-Obstruction / pathology
  • Italy
  • Magnetic Resonance Imaging
  • Male
  • Mitochondrial Encephalomyopathies / genetics*
  • Mitochondrial Encephalomyopathies / pathology
  • Muscular Dystrophy, Oculopharyngeal
  • Mutation / genetics*
  • Ophthalmoplegia / congenital
  • Siblings*
  • Thymidine Phosphorylase / genetics*

Substances

  • Thymidine Phosphorylase

Supplementary concepts

  • Visceral myopathy familial external ophthalmoplegia