Abstract
Chronic graft-versus-host disease (GVHD) is a common long-term complication of allogeneic hematopoietic stem-cell transplantation (HSCT), and is responsible for morbidity, mortality and a decrease in quality of life of patients after SCT. Polymyositis, which usually co-occurs with other manifestations of GVHD, has previously been reported. However, polymyositis as the sole manifestation of chronic GVHD following donor lymphocyte infusion (DLI) is rare. We report a 30-year-old man with Hodgkin’s lymphoma who developed acute polymyositis following treatment by DLI 4 months post-allogeneic HSCT. The patient developed fever and generalized myalgia 22 days after a single dose of DLI. Laboratory testing showed elevated muscle enzymes and myopathic abnormalities on electromyographic examination. Muscle biopsy showed features of acute polymyositis, with widespread foci of muscle fiber necrosis associated with infiltration of small mononuclear cells. Twenty-four hours after diagnosis, the patient developed a fatal ventricular arrhythmia. Cardiac involvement may occur in association with polymyositis, but usually occurs in elderly patients after several months of illness. The present case highlights the importance of systematic cardiac evaluation when a diagnosis of polymyositis is initially made to exclude this infrequent presentation of chronic GVHD characteristically associated with some HLA-DR haplotypes.
References
Ahn JS, Cho SH, Kim YK, Yang DH, Bae WK, Shim HJ, Lee JJ, Chung IJ, Juhng SW, Kim HJ. Polymyositis and myocarditis after donor lymphocyte infusion. Int. J. Hematol. 2009;90:113–6.
Lin PC, Hsiao LT, Chen PM. Acute polymyositis after donor lymphocyte infusion. Eur J Haematol. 2005;74:166–8.
Couriel DR, Beguelin GZ, Giralt S, De Lima M, Hosing C, Kharfan-Dabaja MA, Anagnostopoulos A, Champlin R. Chronic graft-versus-host disease manifesting as polymyositis: an uncommon presentation. Bone Marrow Transplant. 2002;30:543–6.
Goldstein R, Duvic M, Targoff IN, Reichlin M, McMenemy AM, Reveille JD, Warner NB, Pollack MS, Arnett FC. HLA-D region genes associated with autoantibody responses to histidyl-transfer RNA synthetase (Jo-1) and other translation-related factors in myositis. Arthritis Rheum. 1990;33(8):1240–8.
Liu FC, Chen CH, Chao TY. Polymyositis complicating donor lymphocyte infusion after stem cell transplantation for relapsed chronic myeloid leukemia: report of a case and review of literature. Clin Rheumatol. 2007;26(7):1207–10.
Maillard-Lefebvre H, Morell-Dubois S, Lambert M, Charlanne H, Launay D, Hachulla E, Yakoub-Agh I, Hatron PY. Graft-versus-host disease-related polymyositis. Clin Rheumatol. 2010;29(4):431–3.
Bhan A. Fatal myocarditis with acute polymyositis in a young adult. Postgrad Med J. 1990;66(773):229–31.
Rackley C, Schultz KR, Goldman FD, Chan KW, Serrano A, Hulse JE, Gilman AL. Cardiac manifestations of graft-versus-host disease. Biol Blood Marrow Transplant. 2005;11(10):773–80.
Sénéchal M, Crête M, Couture C, Poirier P. Myocardial dysfunction in polymyositis. Can J Cardiol. 2006;22(10):869–71.
Author information
Authors and Affiliations
Corresponding author
About this article
Cite this article
Montoro, J., Hernández-Boluda, J.C., Arbona, C. et al. Polymyositis after donor lymphocyte infusion. Int J Hematol 96, 386–389 (2012). https://doi.org/10.1007/s12185-012-1144-4
Received:
Revised:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s12185-012-1144-4