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Dermatomyositis developed in a recipient of allogeneic BMT; the differentiation of chronic GVHD and autoimmune disease

Bone Marrow Transplantation volume 47, pages 603–604 (2012)Cite this article

Dermatomyositis-like features occurring after allogeneic BMT were thought to be a rare presentation of chronic GVHD (cGVHD).1, 2, 3, 4 Herein, we report a male patient who developed dermatomyositis 14 years after allogeneic BMT. This 24-year-old man was admitted to our hospital with myalgia and muscle weakness in his upper and lower extremities. Examination of his medical history revealed that he had received an allogeneic BMT from an HLA-matched unrelated female donor as a rescue for relapsed ALL at the age of 10 years. The transplant was performed with a conditioning regimen of BU, CY and etoposide with a GVHD prophylaxis of CsA and short-term MTX. A complete engraftment was obtained on day 16. Genotyping using XY-FISH analysis of a BM sample taken at day 26 revealed that 98.8% of whole nucleated cells were of donor origin, suggesting that complete chimera was achieved. The post-transplant course was smooth and uneventful with grade 1 acute GVHD of the skin. Since day 368, he has been free from immunosuppressants for over a decade.

Both polymyositis and dermatomyositis can occur after BMT.5 Dermatomyositis shows skin manifestations including a heliotrope rash and Gottron's papules.6, 7 However, cGVHD-related muscle complications of fascilitis/myositis are very rare. We found only six cases of BMT-related dermatomyositis in the literature,1, 2, 3, 4 which developed at 4–52 months after transplantation. Of a total of seven cases including ours, patients were aged 24–51 years, four were female, and five were diagnosed histopathologically. However, only ours was characterized for the immunophenotypes and genotyping of infiltrating mononuclear cells, which showed predominantly recipient-type CD4+ T-cell infiltration, compatible with the findings of dermatomyositis.6, 7 However, in cGVHD-related diseases, interactions between donor CD8+ cells and host cells must take place, which lead to the inflammatory disease.

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Acknowledgements

We thank Dr Shinsaku Imashuku for his critical reading of the manuscript.

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Authors and Affiliations

  1. Department of Pediatrics, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, Japan

    K Sakamoto, T Imamura & H Hosoi

  2. Department of Neurology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, Japan

    F Niwa & K Shiga

  3. Department of Dermatology, Graduate School of Medical Science, Kyoto Prefectural University of Medicine, Kyoto, Japan

    S Komori

  4. Department of Pediatrics, Fukui Aiiku Hospital, Fukui, Japan

    Y Ishihara

  5. Department of Pathology, Nagoya First Red Cross Hospital, Nagoya, Japan

    M Ito

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Correspondence to T Imamura.

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Sakamoto, K., Imamura, T., Niwa, F. et al. Dermatomyositis developed in a recipient of allogeneic BMT; the differentiation of chronic GVHD and autoimmune disease. Bone Marrow Transplant 47, 603–604 (2012). https://doi.org/10.1038/bmt.2011.125

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