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Trans-vivo Delayed Type Hypersensitivity Assay for Antigen Specific Regulation
11:49

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Published on: May 2, 2013

Polymyositis after donor lymphocyte infusion.

Juan Montoro1, Juan Carlos Hernández-Boluda, Cristina Arbona

  • 1Hematology Department, Hospital Clínico Universitario, Valencia, Spain. juanmontorogomez@hotmail.com

International Journal of Hematology
|August 21, 2012
PubMed
Summary

Polymyositis as the only sign of chronic graft-versus-host disease after donor lymphocyte infusion is rare. Early cardiac evaluation is crucial due to potential fatal arrhythmias.

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Area of Science:

  • Hematology
  • Immunology
  • Neurology

Background:

  • Allogeneic hematopoietic stem-cell transplantation (HSCT) can lead to chronic graft-versus-host disease (GVHD), a major cause of morbidity and mortality.
  • Polymyositis is a known complication of GVHD, but its presentation as the sole manifestation after donor lymphocyte infusion (DLI) is uncommon.

Observation:

  • A 30-year-old man with Hodgkin's lymphoma developed acute polymyositis 4 months post-HSCT following DLI.
  • Symptoms included fever, myalgia, elevated muscle enzymes, and electromyographic abnormalities.
  • Muscle biopsy confirmed polymyositis with muscle fiber necrosis and mononuclear cell infiltration.

Findings:

  • The patient experienced a fatal ventricular arrhythmia 24 hours after polymyositis diagnosis.
  • This case highlights a rare, rapidly progressing presentation of chronic GVHD.
  • Cardiac involvement in polymyositis, though infrequent and typically later-onset, can be fatal.

Implications:

  • Systematic cardiac evaluation is essential upon diagnosing polymyositis in HSCT patients.
  • Early detection of cardiac involvement may prevent fatal arrhythmias.
  • Understanding HLA-DR haplotype associations could refine risk assessment for this rare GVHD manifestation.