Investigation into thiol conjugation of transthyretin in hereditary transthyretin amyloidosis

Eur J Clin Invest. 1998 Aug;28(8):687-92. doi: 10.1046/j.1365-2362.1998.00345.x.

Abstract

Background: For all forms of amyloidosis, the amyloid-generating mechanism is unknown. Familial amyloidotic polyneuropathy type I is caused by a variant transthyretin (TTR Met-30). As electrospray ionization mass spectrometry (ESI-MS) discloses both thiol-conjugated and -unconjugated forms of wild-type and variant TTR, we wanted to investigate the relationship between TTR conjugation and clinically overt amyloid disease.

Methods: Plasma from 35 individuals (12 symptomatic TTR Met-30 carriers, nine asymptomatic and 14 healthy control subjects) were analysed using ESI-MS.

Results: The total TTR concentration was significantly lower in symptomatic TTR Met-30 carriers than in control subjects. An increased percentage of conjugated TTR Met-30 was found in symptomatic carriers compared with asymptomatic, whereas the percentage conjugated wild-type TTR was similar for control subjects, asymptomatic and symptomatic TTR Met-30 carriers.

Conclusion: The finding of a decreased ratio of unconjugated to conjugated TTR Met-30 in plasma samples from symptomatic TTR Met-30 carriers indicates that thiol conjugation of TTR could be involved in amyloid formation.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Amyloid Neuropathies / genetics
  • Amyloid Neuropathies / metabolism*
  • Female
  • Humans
  • Male
  • Mass Spectrometry / methods
  • Metabolism, Inborn Errors / genetics
  • Metabolism, Inborn Errors / metabolism
  • Middle Aged
  • Molecular Weight
  • Point Mutation
  • Prealbumin / chemistry
  • Prealbumin / genetics
  • Prealbumin / metabolism*
  • Sulfhydryl Compounds / chemistry
  • Sulfhydryl Compounds / metabolism*

Substances

  • Prealbumin
  • Sulfhydryl Compounds