Journal of the American Academy of Dermatology
Ruzicka et al. lesions after 1 week. Histologic examination showed findings suggestive of SCLE and negative DIF analogous to those described by Lehmann et aL J0 for UV-induced SCLE lesions.
REFERENCES 1. Teramoto N, Katayama I, Arai H, et al. Annular erythema: a possible association with primary Sjogren's syndrome. J AM ACAD DERMATOL 1989;20:596-601. 2. Katayama I, Asai T, Nishioka K, et al. Annular erythema associated with primary Sjogren's syndrome: analysis of T cell subsets in cutaneous infiltrates. JAM ACAD DERMATOL 1989;21:1218-21. 3. Tan EN, Cohen AS, Fries JF, et al. The 1982 revised criteria for the classification of systemic lupus erythematosus. Arthritis Rheum 1982;25:1271-7. 4. Sontheimer RD, Thomas JR, Gilliam IN. Subacute cutaneous lupus erythematosus-a cutaneous marker for a distinct LE subset. Arch Dermatol 1979;115:1409-15.
5. Sontheimer RD, Maddison PJ, Reichlin M, et al. Serologic and HLA associations in subacute cutaneous lupus erythematosus, a clinical subset of lupus erythematosus. Ann Intern Med 1982;97:664-71. 6. Sontheimer RD, Stastny P, Gilliam IN. Human histocompatibility antigen associations in subacute cutaneous lupus erythematosus. J Clin Invest 1981;67:313-6. 7. Sontheimer RD. Immunobiological significance of the RoSSA antigen-antibody system. Arch Dermatol 1985; 121:327-9. 8. Rekant SI, Becker LE. Auto-immune annular erythema. A variant of lupus erythematosus? Arch Dermatol 1973; 107:424-6. 9. Provost TT, Talal N, Harley JB, et aI. The relationship between anti-Ro (SS-A) antibody-positive Sjogren's syndrome and anti-Ro (SS-A) antibody-positive lupus erythematosus. Arch Dermatol 1988;124:62-71. 10. Lehmann P, HOIzle E, Kind P, et al. Experimental reproduction of skin lesions in lupus erythematosus by UVA anli UVB radiation. JAM ACAD DERMATOL 1990;22:181-7.
Polymyositis: A manifestation of chronic graftversus-host disease R. Prussick, MD, M. C. Brain, DM, FRCP, FRCPC, 1. R. Walker, MBBS, FRCPC, and D. N. Sauder, MD, FRCPC Hamilton, Ontario, Canada Polymyositis developed in a patient who had had bone marrow transplants for the treatment of acute myeloid leukemia. There was no previous evidence ofgraft-versus-host disease. Polymyositis has previously been reported to be associated with graft-versus-host disease; this article suggests that polymyositis may represent its sole manifestation. (J AM ACAD DERMATOL 1991;25:560-2.)
A major complication of bone marrow transplantation is graft-versus-host disease (GVHD), the major manifestations of which include cutaneous changes, hepatic impairment, and gastrointestinal involvement. Chronic GVHD, which may occur with or without preceding acute forms, shows many clinical and pathologic similarities to autoimmune diseases such as progressive systemic sclerosis and From the Department of Medicine, Division of Dermatology and Hematology. McMaster University. Reprint requests: Daniel N. Sauder, MD, Division of Dennato]ogy, University of Toronto, Sunnybrook Health Science Centre, Toronto, Ontario, Canada M4N 3MS.
16/4/24608
560
Sjogren's syndrome. In the early 1980s polymyositis was reported to be associated with chronic GVHD. The majority of cases have also been associated with the skin changes of chronic GVHD. We report the case of a patient in whom, 3 years after bone marrow transplantation, polymyositis associated with the sicca syndrome developed. This case supports the concept that polymyositis may be the only manifestation of chronic GVHD. CASE REPORT
In December 1986 a diagnosis of acute myeloid leukemia subtype M.I.! was established in a 29-year-old man. The patient was treated with daunorubicin and cytosine arabinoside. In February 1987, he was admitted for his
Volume 25 Number 3 September 1991
Polymyositis in chronic graft-versus-host disease 561
,
, ,
Fig. 1. Muscle biopsy specimen from deltoid reveals perivascular and perifascicular lymphocytic infiltration compatible with polymyositis. (Hematoxylin-eosin stain; X132.) first course of consolidation chemotherapy (3 days of daunorubicin and 7 days of cytosine arabinoside). In March 1987, the patient was readmitted because of febrile neutropenia, Streptococcus fecalis septicemia, and thrombocytopenia. He completed only one of three intended courses of consolidation chemotherapy because of persistent thrombocytopenia and intermittent leukopenia. In October 1987, the patient received an allogeneic bone marrOw transplant from an HLA-matched brother (mixed lymphocyte culture-nonreactive). Engraftment was prompt, with neutrophils first appearing on day 10. Skin biopsy specimens of several small erythematous papules on the extensor surface of his forearms that were first noted 10 days after the bone marrow transplantation showed rare necrotic keratinocytes within the epidermis. There was no significant inflammation or liquefaction degeneration at the dermoepidermal junction. This was compatible with the cytotoxic effect of his medications and radiation treatment; there was no evidence ofGVHD. No lesions of the skin or mucous membranes were found elsewhere. During the next few months he noted progressively worsening dry eyes and mouth. In September 1988, approximately 11 months after the transplantation, the patient noticed progressive muscle weakness, especially in his thighs. Physical examination revealed only muscle weakness, particularly of the proximal muscles. In January 1989, although an electromyogram was not diagnostic, muscle biopsy specimens of deltoid and vastus laterali~ showed a perivascular and perifascicular lymphocytic infiltrate consistent with an inflammatory myopathy without marked fiber atrophy or vasculitis (Fig. 1). A skin biopsy specimen revealed no evidence of GVHD. Creatinine kinase was normal, but aldolase was elevated
at 9UjL (normal
Ruzicka et al. lesions after 1 week. Histologic examination showed findings suggestive of SCLE and negative DIF analogous to those described by Lehmann et aL J0 for UV-induced SCLE lesions.
REFERENCES 1. Teramoto N, Katayama I, Arai H, et al. Annular erythema: a possible association with primary Sjogren's syndrome. J AM ACAD DERMATOL 1989;20:596-601. 2. Katayama I, Asai T, Nishioka K, et al. Annular erythema associated with primary Sjogren's syndrome: analysis of T cell subsets in cutaneous infiltrates. JAM ACAD DERMATOL 1989;21:1218-21. 3. Tan EN, Cohen AS, Fries JF, et al. The 1982 revised criteria for the classification of systemic lupus erythematosus. Arthritis Rheum 1982;25:1271-7. 4. Sontheimer RD, Thomas JR, Gilliam IN. Subacute cutaneous lupus erythematosus-a cutaneous marker for a distinct LE subset. Arch Dermatol 1979;115:1409-15.
5. Sontheimer RD, Maddison PJ, Reichlin M, et al. Serologic and HLA associations in subacute cutaneous lupus erythematosus, a clinical subset of lupus erythematosus. Ann Intern Med 1982;97:664-71. 6. Sontheimer RD, Stastny P, Gilliam IN. Human histocompatibility antigen associations in subacute cutaneous lupus erythematosus. J Clin Invest 1981;67:313-6. 7. Sontheimer RD. Immunobiological significance of the RoSSA antigen-antibody system. Arch Dermatol 1985; 121:327-9. 8. Rekant SI, Becker LE. Auto-immune annular erythema. A variant of lupus erythematosus? Arch Dermatol 1973; 107:424-6. 9. Provost TT, Talal N, Harley JB, et aI. The relationship between anti-Ro (SS-A) antibody-positive Sjogren's syndrome and anti-Ro (SS-A) antibody-positive lupus erythematosus. Arch Dermatol 1988;124:62-71. 10. Lehmann P, HOIzle E, Kind P, et al. Experimental reproduction of skin lesions in lupus erythematosus by UVA anli UVB radiation. JAM ACAD DERMATOL 1990;22:181-7.
Polymyositis: A manifestation of chronic graftversus-host disease R. Prussick, MD, M. C. Brain, DM, FRCP, FRCPC, 1. R. Walker, MBBS, FRCPC, and D. N. Sauder, MD, FRCPC Hamilton, Ontario, Canada Polymyositis developed in a patient who had had bone marrow transplants for the treatment of acute myeloid leukemia. There was no previous evidence ofgraft-versus-host disease. Polymyositis has previously been reported to be associated with graft-versus-host disease; this article suggests that polymyositis may represent its sole manifestation. (J AM ACAD DERMATOL 1991;25:560-2.)
A major complication of bone marrow transplantation is graft-versus-host disease (GVHD), the major manifestations of which include cutaneous changes, hepatic impairment, and gastrointestinal involvement. Chronic GVHD, which may occur with or without preceding acute forms, shows many clinical and pathologic similarities to autoimmune diseases such as progressive systemic sclerosis and From the Department of Medicine, Division of Dermatology and Hematology. McMaster University. Reprint requests: Daniel N. Sauder, MD, Division of Dennato]ogy, University of Toronto, Sunnybrook Health Science Centre, Toronto, Ontario, Canada M4N 3MS.
16/4/24608
560
Sjogren's syndrome. In the early 1980s polymyositis was reported to be associated with chronic GVHD. The majority of cases have also been associated with the skin changes of chronic GVHD. We report the case of a patient in whom, 3 years after bone marrow transplantation, polymyositis associated with the sicca syndrome developed. This case supports the concept that polymyositis may be the only manifestation of chronic GVHD. CASE REPORT
In December 1986 a diagnosis of acute myeloid leukemia subtype M.I.! was established in a 29-year-old man. The patient was treated with daunorubicin and cytosine arabinoside. In February 1987, he was admitted for his
Volume 25 Number 3 September 1991
Polymyositis in chronic graft-versus-host disease 561
,
, ,
Fig. 1. Muscle biopsy specimen from deltoid reveals perivascular and perifascicular lymphocytic infiltration compatible with polymyositis. (Hematoxylin-eosin stain; X132.) first course of consolidation chemotherapy (3 days of daunorubicin and 7 days of cytosine arabinoside). In March 1987, the patient was readmitted because of febrile neutropenia, Streptococcus fecalis septicemia, and thrombocytopenia. He completed only one of three intended courses of consolidation chemotherapy because of persistent thrombocytopenia and intermittent leukopenia. In October 1987, the patient received an allogeneic bone marrOw transplant from an HLA-matched brother (mixed lymphocyte culture-nonreactive). Engraftment was prompt, with neutrophils first appearing on day 10. Skin biopsy specimens of several small erythematous papules on the extensor surface of his forearms that were first noted 10 days after the bone marrow transplantation showed rare necrotic keratinocytes within the epidermis. There was no significant inflammation or liquefaction degeneration at the dermoepidermal junction. This was compatible with the cytotoxic effect of his medications and radiation treatment; there was no evidence ofGVHD. No lesions of the skin or mucous membranes were found elsewhere. During the next few months he noted progressively worsening dry eyes and mouth. In September 1988, approximately 11 months after the transplantation, the patient noticed progressive muscle weakness, especially in his thighs. Physical examination revealed only muscle weakness, particularly of the proximal muscles. In January 1989, although an electromyogram was not diagnostic, muscle biopsy specimens of deltoid and vastus laterali~ showed a perivascular and perifascicular lymphocytic infiltrate consistent with an inflammatory myopathy without marked fiber atrophy or vasculitis (Fig. 1). A skin biopsy specimen revealed no evidence of GVHD. Creatinine kinase was normal, but aldolase was elevated
at 9UjL (normal
