Development of autoimmune exocrinopathy resembling Sjögren's syndrome in estrogen-deficient mice of healthy background

Am J Pathol. 2003 Oct;163(4):1481-90. doi: 10.1016/S0002-9440(10)63505-5.

Abstract

Although a number of autoimmune diseases are known to develop in postmenopausal women, the mechanisms by which estrogen deficiency influences autoimmune lesions remain unclear. We speculate that antiestrogenic actions might be a potent factor in the formation of pathogenic autoantigens. Previously, we have identified 120-kd alpha-fodrin as an important autoantigen in Sjögren's syndrome (SS). When healthy C57BL/6 (B6) mice were treated with an ovariectomy (Ovx), we found a significant increase in TUNEL(+)-apoptotic epithelial cells in the salivary gland cells associated with alpha-fodrin cleavage during 2 and 3 weeks after Ovx. By contrast, no apoptotic cells were found in estrogen receptor-alpha knockout mice. In in vitro studies using primary cultured mouse salivary gland cells and human salivary gland cells, we found a cleavage product of 120-kd alpha-fodrin in cells that had undergone tamoxifen (Tam)-induced apoptosis through caspase activation, especially caspase-1. Adoptive transfer of alpha-fodrin-reactive T cells into Ovx-B6 and -SCID mice resulted in the development of autoimmune exocrinopathy quite similar to SS. These results suggest that estrogen deficiency exerts a crucial influence on autoantigen cleavage, and may cause, in part, autoimmune exocrinopathy in postmenopausal women.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adoptive Transfer
  • Animals
  • Apoptosis
  • Carrier Proteins / chemistry
  • Carrier Proteins / immunology
  • Carrier Proteins / metabolism
  • Caspases / metabolism
  • Estrogen Receptor alpha
  • Estrogens / deficiency*
  • Female
  • Humans
  • Mice
  • Mice, Inbred C57BL
  • Mice, Knockout
  • Mice, SCID
  • Microfilament Proteins / chemistry
  • Microfilament Proteins / immunology
  • Microfilament Proteins / metabolism
  • Ovariectomy
  • Peptide Hydrolases / metabolism
  • Receptors, Estrogen / deficiency
  • Salivary Gland Diseases / etiology*
  • Salivary Gland Diseases / pathology
  • Salivary Gland Diseases / physiopathology*
  • Salivary Glands / pathology
  • Salivary Glands / physiopathology
  • Sjogren's Syndrome / pathology
  • Sjogren's Syndrome / physiopathology*
  • T-Lymphocytes / immunology
  • Tamoxifen / pharmacology
  • Tumor Cells, Cultured

Substances

  • Carrier Proteins
  • Estrogen Receptor alpha
  • Estrogens
  • Microfilament Proteins
  • Receptors, Estrogen
  • fodrin
  • Tamoxifen
  • Peptide Hydrolases
  • Caspases