Disease specificity of anti-tryptophan hydroxylase-1 and anti-AIE-75 autoantibodies in APECED and IPEX syndrome

Clin Immunol. 2015 Jan;156(1):36-42. doi: 10.1016/j.clim.2014.10.010. Epub 2014 Nov 8.

Abstract

Autoantibodies to autoimmune enteropathy-related 75 kDa antigen (AIE-75) and villin are disease markers of immune dysregulation, polyendocrinopathy, enteropathy, X-linked (IPEX) syndrome which is characterized by a peripheral tolerance defect. On the other hand, anti-tryptophan hydroxylase-1 (TPH-1) antibodies are detected in autoimmune polyendocrinopathy, candidiasis, ectodermal dystrophy (APECED), a central tolerance defect, especially when complicated with gastrointestinal dysfunction. However, to date, anti-AIE-75 and anti-villin antibodies or anti-TPH-1 antibodies have not been tested in APECED or IPEX syndrome, respectively. In the present study, we confirmed the disease specificity of both anti-AIE-75 and anti-TPH-1, although anti-villin antibodies were detected in some patients with APECED. Our observation suggests that immunotolerance to AIE-75 depends on the peripheral mechanism, whereas the tolerance to TPH-1 depends on the central mechanisms.

Keywords: AIE-75;; APECED;; IPEX syndrome; Tryptophan hydroxylase;; Villin;.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adaptor Proteins, Signal Transducing / metabolism*
  • Autoantibodies / blood*
  • Cell Cycle Proteins
  • Cytoskeletal Proteins
  • Diabetes Mellitus, Type 1 / congenital
  • Diagnosis, Differential
  • Diarrhea
  • Genetic Diseases, X-Linked / diagnosis
  • Genetic Diseases, X-Linked / immunology
  • Immune System Diseases / congenital
  • Immune Tolerance
  • Immunoblotting
  • Polyendocrinopathies, Autoimmune / diagnosis
  • Polyendocrinopathies, Autoimmune / immunology*
  • Tryptophan Hydroxylase / metabolism*

Substances

  • Adaptor Proteins, Signal Transducing
  • Autoantibodies
  • Cell Cycle Proteins
  • Cytoskeletal Proteins
  • USH1C protein, human
  • TPH1 protein, human
  • Tryptophan Hydroxylase

Supplementary concepts

  • Immune Dysregulation, Polyendocrinopathy, Enteropathy, X-Linked Syndrome