Diffuse pruritic erythema as a clinical manifestation in anti-SAE antibody-associated dermatomyositis: a case report and literature review

Clin Rheumatol. 2019 Aug;38(8):2189-2193. doi: 10.1007/s10067-019-04562-w. Epub 2019 Apr 27.

Abstract

Anti-small ubiquitin-like modifier-1 activating enzyme (anti-SAE) antibodies have been recently discovered especially for myosin and identified as dermatomyositis (DM) marker. The frequency of anti-SAE antibodies in DM patients is extremely low. Diffuse pruritic erythema may be one kind of clinical manifestations of DM with anti-SAE antibodies. In this report, a 48-year-old female patient with amyopathic dermatomyositis (ADM) carrying anti-SAE antibodies presented diffuse pruritic erythema for 5 months. Diffuse pruritic erythema improved after treatment with prednisolone, cyclosporine, and thalidomide. The clinical characteristics of 75 previously reported cases with anti-SAE antibody-positive DM were reviewed, and the manifestations of the Asian and Western cohorts were compared. It was revealed that the Asian patients were more susceptible to diffuse erythema (17/34 vs. 3/41, P = 0.000), dysphagia (16/34 vs. 10/41, P = 0.040), and interstitial lung disease (ILD) (21/34 vs. 5/41, P = 0.000) compared with the Western patients. The frequency of malignancy in the Asian cohort was significantly higher than that in the Western cohort (10/34 vs. 4/41, P = 0.030).

Keywords: Anti-SAE antibody; Anti-small ubiquitin-like modifier-1 activating enzyme antibody; Dermatomyositis; Diffuse erythema; Literature review; Malignancy.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Antibodies / immunology
  • Asian People
  • Cyclosporine / administration & dosage
  • Dermatomyositis / complications*
  • Dermatomyositis / ethnology
  • Erythema / complications*
  • Erythema / ethnology
  • Female
  • Humans
  • Methotrexate / administration & dosage
  • Middle Aged
  • Prednisolone / administration & dosage
  • Pruritus / complications*
  • Pruritus / ethnology
  • Thalidomide / administration & dosage
  • Treatment Outcome
  • Ubiquitin-Activating Enzymes / immunology*

Substances

  • Antibodies
  • Thalidomide
  • Cyclosporine
  • Prednisolone
  • SAE1 protein, human
  • Ubiquitin-Activating Enzymes
  • Methotrexate

Supplementary concepts

  • Amyopathic dermatomyositis