Dialysis-related amyloidosis associated with a novel β2-microglobulin variant

Amyloid. 2021 Mar;28(1):42-49. doi: 10.1080/13506129.2020.1813097. Epub 2020 Sep 2.

Abstract

Till date, there had been no reported case of dialysis-related amyloidosis (DRA) associated with a β2-microglobulin variant. We report here a 41-year-old haemodialysis patient with systemic amyloidosis, exhibiting macroglossia and swelling salivary glands, uncommon clinical manifestations for DRA. Molecular analysis showed that the patient had a new variant of β2-microglobulin (V27M). Extracted amyloid protein was predominantly composed of variant β2-microglobulin. In vitro analysis revealed that this variant β2-microglobulin had a strong amyloidogenic propensity, probably owing to the decreased stability caused by a bulky methionine residue. Our data clearly show that V27M variant is amyloidogenic and this mutation results in unusual clinical manifestations. To date, only one amyloidogenic β2-microglobulin variant (D76N) has been reported in non-dialysis patients. It is noteworthy that the V27M and D76N variants show substantial differences in both clinical phenotypes and pathomechanical features. This is the first case of DRA associated with a naturally occurring β2-microglobulin variant.

Keywords: Dialysis-related amyloidosis; disulphide bond; variant β2-microglobulin; wild-type β2-microglobulin.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Amino Acid Substitution / genetics
  • Amyloid / genetics*
  • Amyloidosis / etiology
  • Amyloidosis / genetics*
  • Amyloidosis / pathology
  • Humans
  • Male
  • Mutation, Missense / genetics
  • Protein Aggregation, Pathological / genetics*
  • Renal Dialysis / adverse effects
  • beta 2-Microglobulin / genetics*

Substances

  • Amyloid
  • beta 2-Microglobulin