Loss of gastric interstitial cells of Cajal in patients with hereditary transthyretin amyloidosis

Amyloid. 2013 Jun;20(2):99-106. doi: 10.3109/13506129.2013.787985. Epub 2013 May 3.

Abstract

Background: Hereditary transthyretin (TTR) amyloidosis is a systemic neuropathic disorder caused by TTR gene mutations. Gastrointestinal complications are common and the underlying mechanisms remain unclear. The interstitial cells of Cajal (ICC) function as pacemaker cells in the gastrointestinal tract and are important for gastrointestinal motility. The aim of this study was to investigate the densities of gastric ICC and nerves in patients with TTR amyloidosis compared to non-amyloidosis controls.

Methods: Antral wall autopsy specimens from 11 Japanese ATTR V30M patients and 10 controls were analyzed with immunohistochemistry and computerized analysis. Antibodies to c-Kit and TMEM16A were used to assess ICC and an antibody to PGP 9.5 was used to assess nervous tissue. The study was approved by a Japanese ethical committee.

Results: The densities of c-Kit-immunoreactive (IR) ICC were significantly lower in the circular and longitudinal muscle layers of patients compared to controls (p = 0.004 for both). Equivalent results were found for TMEM16A-IR ICC. There were no significant differences in PGP 9.5-IR cells in the circular or longitudinal muscle layers between patients and controls (p = 0.173 and 0.099, respectively).

Conclusions: A loss of gastrointestinal ICC may be an important factor for the digestive disturbances in hereditary TTR amyloidosis.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Amyloid / metabolism*
  • Amyloid Neuropathies, Familial / metabolism
  • Amyloid Neuropathies, Familial / pathology*
  • Anoctamin-1
  • Case-Control Studies
  • Chloride Channels / metabolism
  • Female
  • Gastric Mucosa / metabolism
  • Humans
  • Image Processing, Computer-Assisted
  • Immunoenzyme Techniques
  • Interstitial Cells of Cajal / metabolism
  • Interstitial Cells of Cajal / pathology*
  • Male
  • Middle Aged
  • Muscle, Smooth / metabolism
  • Muscle, Smooth / pathology
  • Neoplasm Proteins / metabolism
  • Nervous System / metabolism
  • Nervous System / pathology
  • Prealbumin / metabolism*
  • Proto-Oncogene Proteins c-kit / metabolism
  • Sex Factors
  • Stomach / pathology*

Substances

  • ANO1 protein, human
  • Amyloid
  • Anoctamin-1
  • Chloride Channels
  • Neoplasm Proteins
  • Prealbumin
  • Proto-Oncogene Proteins c-kit

Supplementary concepts

  • Amyloidosis, Hereditary, Transthyretin-Related