"Borrelia-associated early-onset morphea": a particular type of scleroderma in childhood and adolescence with high titer antinuclear antibodies? Results of a cohort analysis and presentation of three cases

J Am Acad Dermatol. 2009 Feb;60(2):248-55. doi: 10.1016/j.jaad.2008.09.023. Epub 2008 Nov 20.

Abstract

Background: Morphea is an inflammatory autoimmune skin sclerosis of unknown etiology. A causative role of Borrelia burgdorferi infection has been controversially discussed, but no conclusive solution has yet been achieved.

Objective: Intrigued by 3 young patients with severe Borrelia-associated morphea and high-titer antinuclear antibodies, we retrospectively examined the relationship between Borrelia exposure, serologic autoimmune phenomena and age at disease onset in morphea patients.

Methods: In 90 morphea patients the presence of Borrelia-specific serum antibodies was correlated to the age at disease onset and the presence and titers of antinuclear antibodies. Patients with active Borrelia infection or high-titer antinuclear antibodies due to systemic sclerosis or lupus erythematosus served as controls.

Results: We observed a statistically highly significant association between morphea, serologic evidence of Borrelia infection, and high-titer antinuclear antibodies when disease onset was in childhood or adolescence.

Limitations: Because pathogenic Borrelia species may vary in different geographic regions the relevance of Borrelia infection in morphea induction may show regional variations.

Conclusion: B burgdorferi infection may be relevant for the induction of a distinct autoimmune type of scleroderma; it may be called "Borrelia-associated early onset morphea" and is characterized by the combination of disease onset at younger age, infection with B burgdorferi, and evident autoimmune phenomena as reflected by high-titer antinuclear antibodies. As exemplified by the case reports, it may take a particularly severe course and require treatment of both infection and skin inflammation.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Age of Onset
  • Aged
  • Aged, 80 and over
  • Antibodies, Antinuclear / blood*
  • Antibodies, Bacterial / blood
  • Antibody Specificity
  • Borrelia burgdorferi*
  • Child
  • Cohort Studies
  • Female
  • Humans
  • Immunoglobulin G / blood
  • Immunoglobulin M / blood
  • Lyme Disease / complications*
  • Lyme Disease / immunology*
  • Male
  • Middle Aged
  • ROC Curve
  • Retrospective Studies
  • Scleroderma, Localized / immunology*
  • Scleroderma, Localized / microbiology*
  • Young Adult

Substances

  • Antibodies, Antinuclear
  • Antibodies, Bacterial
  • Immunoglobulin G
  • Immunoglobulin M