Lewy body pathology in familial Alzheimer disease: evidence for disease- and mutation-specific pathologic phenotype

Arch Neurol. 2006 Mar;63(3):370-6. doi: 10.1001/archneur.63.3.370.

Abstract

Background: The origin and significance of Lewy bodies and neurites (Lewy body pathology [LBP]) in Alzheimer disease (AD) are poorly understood.

Objective: To examine LBP in the brainstem, limbic cortex, and neocortex of a large number of familial AD cases with mutations in 2 presenilin (PSEN) genes.

Methods: Twenty-five familial AD cases with 9 known PSEN 1 mutations and 14 familial AD cases with a single PSEN 2 mutation (N141I) were examined for LBP using alpha-synuclein immunohistochemistry and sampling of multiple brainstem and cortical regions.

Results: The amygdala was the most vulnerable site for LBP. In fact, virtually all (24 [96%] of 25 cases) of the PSEN 1 mutation cases had LBP in the amygdala. The PSEN 1 mutation cases also had more frequent LBP in the amygdala and neocortex than those with the PSEN 2 mutation. However, within families with a single mutation of either PSEN 1 or PSEN 2, there was frequent variability of the LBP.

Conclusion: These findings suggest that there are genetic influences on the presence of LBP in familial AD as demonstrated by the differences between PSEN 1 and PSEN 2 mutation cases.

Publication types

  • Comparative Study

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Alzheimer Disease / genetics*
  • Alzheimer Disease / pathology*
  • Apolipoproteins E / genetics
  • Brain / metabolism
  • Brain / pathology
  • Female
  • Genotype
  • Humans
  • Immunohistochemistry / methods
  • Lewy Bodies / genetics
  • Lewy Bodies / pathology*
  • Male
  • Membrane Proteins / genetics
  • Middle Aged
  • Mutation
  • Neurites / pathology
  • Neuropsychological Tests / statistics & numerical data
  • Presenilin-1
  • Presenilin-2
  • alpha-Synuclein / metabolism

Substances

  • Apolipoproteins E
  • Membrane Proteins
  • PSEN1 protein, human
  • PSEN2 protein, human
  • Presenilin-1
  • Presenilin-2
  • SNCA protein, human
  • alpha-Synuclein