Recurrence of anti-MDA5 antibody-positive clinically amyopathic dermatomyositis after long-term remission: A case report

Medicine (Baltimore). 2018 Jun;97(26):e11024. doi: 10.1097/MD.0000000000011024.

Abstract

Rationale: Among all dermatomyositis (DM) patients, antimelanoma differentiation-associated gene 5 antibody (anti-MDA5 Ab) positive patients have significantly poor short-term mortality, whereas they experience less relapses over the long term after the remission. We report the case of a patient with anti-MDA5 Ab-positive clinically amyopathic dermatomyositis (CADM) with the recurrence of interstitial lung disease (ILD) after 7 years of remission. There has been no case report of an anti-MDA5 Ab-positive DM patient with the recurrence of ILD after 7 years of long-term remission.

Patient concerns: A 70-year-old Japanese woman was diagnosed with anti-MDA5 Ab-positive CADM and ILD. After achieving 7 years long-term remission, she was admitted to our department with erythema on the fingers and interstitial pneumonia. Her anti-MDA5 Ab titer was elevated.

Diagnoses: We diagnosed recurrent CADM complicated with ILD.

Interventions: We successfully treated her with 1,000 mg of methyl-prednisolone pulse and intravenous cyclophosphamide therapy followed by prednisolone 50 mg/day and an increase of cyclosporine.

Outcomes: After that treatment, the patient's skin symptoms and interstitial pneumonia were relieved. All laboratory investigations such as ferritin, the serum markers of interstitial pneumonia (i.e., SP-A, SP-D), and the titer of anti-MDA5 Ab showed signs of improvement.

Lessons: Her case suggests that careful physical examinations and monitoring the serum markers are important even after long-term remission is achieved.

Publication types

  • Case Reports

MeSH terms

  • Administration, Intravenous
  • Aged
  • Anti-Inflammatory Agents / therapeutic use
  • Asian People / ethnology
  • Autoantibodies / blood*
  • Biomarkers / blood
  • Cyclophosphamide / administration & dosage
  • Cyclophosphamide / therapeutic use
  • Cyclosporine / administration & dosage
  • Cyclosporine / therapeutic use
  • Dermatomyositis / complications
  • Dermatomyositis / diagnosis
  • Dermatomyositis / drug therapy
  • Dermatomyositis / immunology*
  • Female
  • Ferritins / blood
  • Ferritins / drug effects
  • Humans
  • Immunosuppressive Agents / therapeutic use
  • Interferon-Induced Helicase, IFIH1 / antagonists & inhibitors*
  • Interferon-Induced Helicase, IFIH1 / immunology
  • Lung Diseases, Interstitial / complications*
  • Lung Diseases, Interstitial / diagnostic imaging
  • Lung Diseases, Interstitial / drug therapy
  • Lung Diseases, Interstitial / immunology
  • Methylprednisolone / administration & dosage
  • Methylprednisolone / therapeutic use
  • Recurrence
  • Tomography, X-Ray Computed
  • Treatment Outcome

Substances

  • Anti-Inflammatory Agents
  • Autoantibodies
  • Biomarkers
  • Immunosuppressive Agents
  • Cyclosporine
  • Cyclophosphamide
  • Ferritins
  • IFIH1 protein, human
  • Interferon-Induced Helicase, IFIH1
  • Methylprednisolone

Supplementary concepts

  • Amyopathic dermatomyositis