Polymyositis in Chronic Graft vs Host Disease

Anderson, Brian A.; Young, P. Vincent; Kean, Walter F.; Ludwin, Samuel K.; Galbraith, Peter R.; Anastassiades, Tassos

doi:10.1001/archneur.1982.00510150058015

Cited by 50 publications

(23 citation statements)

References 12 publications

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“…In 30 of these patients, polymyositis was associated with dermatological changes of GVHD. 2,12,[14][15][16][17][18][19][20][21][22][23][24][25][26] The onset of GVHD-associated polymyositis has been reported to occur between 100 days and 69 months after allogeneic HSCT, and about half of the patients do not have a history of acute GVHD. 21,23 Clinical manifestations of GVHD-associated polymyositis are indistinguishable from those of the idiopathic form polymyositis.…”

Section: Discussionmentioning

confidence: 99%

“…In 16 cases the steroid was used in combination with a variety of immunosuppressants, including: cyclosporine, intravenous immunoglobulin, azathioprine, antilymphocyte globulin, cyclophosphamide, methotrexate, thalidomide and photopheresis. 2,12,[14][15][16][17][18]24,26,27 Thirteen of 30 evaluable patients failed initial immunosuppression and are summarized in Table 1. Six patients were primarily resistant and seven had recurrent polymyositis while steroids were being tapered.…”

Section: Discussionmentioning

confidence: 99%

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Chronic graft-versus-host disease manifesting as polymyositis: an uncommon presentation

Couriel

Beguelin

Giralt

et al. 2002

Bone Marrow Transplant

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Summary:Graft-versus-host disease (GVHD) remains a major complication of allogeneic hematopoietic stem cell transplantation. Polymyositis can occur in association with chronic GVHD and mimics the idiopathic form of the disease. We report two cases of chronic GVHD-associated polymyositis and review the published literature. The two patients presented 13 and 19 months after allogeneic transplantation with characteristic features of muscular hypotrophy, proximal muscle weakness, pain, elevated creatine phosphokinase (CPK), aldolase and SGPT. Interestingly, both patients had HLA DR52 genes, which is frequently reported in association with idiopathic polymyositis. Electromyogram (EMG) and muscle biopsy confirmed the diagnosis. Treatment with cyclosporine or tacrolimus resulted in complete and sustained remission of polymyositis in both cases. A review of the literature shows cyclosporine and steroids are well-described treatment options for patients with myositis in post transplant, as well as idiopathic cases. The duration of immunosuppressive treatment has varied in different reports, and there is a risk of recurrence when immunosuppression is tapered.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Chronic graft-versus-host disease manifesting as polymyositis: an uncommon presentation

Couriel

Beguelin

Giralt

et al. 2002

Bone Marrow Transplant

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“…The disease commonly involves the skin, eyes, mouth, bowel, liver, lung and immune system. 1 The literature reports eight cases of MG 2,4-8 and 29 cases of PM 1,3,[8][9][10][11][12][13][14][15] as unusual manifestations of cGVHD. Our case report is the first reported with both MG and PM developing after allogeneic BMT in a single patient.…”

Section: Discussionmentioning

confidence: 99%

“…1,3,8,9,13 The activity of the cGVHD rather than severity of the myositis determined prognosis. Our patient partially responded to only 2 days of therapy, followed by a prolonged spontaneous recovery phase.…”

Section: Discussionmentioning

confidence: 99%

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Myasthenia gravis and polymyositis as manifestations of chronic graft-versus-host-disease

Saunders

Silverman

et al. 1999

Bone Marrow Transplant

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Summary:Myasthenia gravis and polymyositis are each a rare manifestation of immune dysregulation in chronic graftversus-host disease (cGVHD). We report a 4-year-old boy with idiopathic acquired aplastic anemia who developed myasthenia gravis 22 months and polymyositis 69 months after an allogeneic BMT (5/6 matched, MLC-nonreactive). The occurrence of both syndromes in one patient is unique. Autoimmune dysfunction may be associated with the development of cGVHD as demonstrated by the high incidence of prior aplastic anemia in BMT patients presenting with myasthenia gravis and polymyositis. Recognition of these neurologic manifestations is important in the diagnosis and treatment of cGVHD. Keywords: chronic graft-versus-host disease; myasthenia gravis; polymyositis Chronic graft-versus-host disease (cGVHD) is a complication of allogeneic BMT which is associated with a high degree of morbidity and mortality. cGVHD commonly involves the skin, eyes, mouth, bowel, liver, lung and immune system. 1 Myasthenia gravis (MG) and polymyositis (PM) have been reported as rare manifestations of cGVHD. [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15] This report illustrates a case of MG and PM developing sequentially after an allogeneic BMT in a single patient. Case reportIn December 1990, a 4-year-old boy received an allogeneic BMT from his 5/6 serologically HLA-matched, MLC-nonreactive sister (mismatch A3/29) for treatment of severe idiopathic acquired aplastic anemia. He was conditioned with CY (200 mg/kg) and total body irradiation (300 cGy, single dose). MTX and CsA were given as GVHD prophylaxis. Engraftment of all three lines occurred by day 28 and

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Rehabilitation following bone marrow transplantation

Gillis¹,

Donovan²

2001

Cancer

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Bone marrow transplantation and stem cell transplantation are increasingly used to treat hematologic malignancies and some solid tumors. The treatment entails bone marrow–ablative therapies and intensive medical support to sustain the patient through pancytopenia and other complications of the disease, transplantation process, or drug side effects. Patients who develop graft‐versus‐host disease are the most difficult subset of transplant recipients to manage. Most transplant recipients perform at normal or near‐normal functional levels at the inception of the transplantation process but are at high risk for developing functional deficits as a result of cumulative impairments. These impairments arise from their disease, their prior cancer treatment, transplant induction, graft‐versus‐host disease, immobility, infection, steroid‐related side effects, and other sequelae of transplantation. Preventive and preemptive rehabilitation interventions can minimize functional loss and facilitate recovery, but the transplantation team must be sensitive to and regularly assess for early functional declines in these patients. The physiatrist and the other members of the rehabilitation team must be thoroughly acquainted with the unique needs and challenges of the bone marrow transplantation population in order to design and modify treatment programs effectively and safely. Outcome research has shown that some patients have continued limitations in function despite successful transplantation. Few evidence‐based data are available that addresses factors correlating with poor functional outcomes other than graft‐versus‐host disease. However, this disease has not been investigated utilizing objective functional instruments. Future research should more clearly elucidate the functional impact of allogeneic and autologous transplants by using standardized physical performance measures as well as thorough function‐based symptomatology questionnaires. Cancer 2001;92:998–1007. © 2001 American Cancer Society.

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Polymyositis in Chronic Graft vs Host Disease

Cited by 50 publications

References 12 publications

Chronic graft-versus-host disease manifesting as polymyositis: an uncommon presentation

Chronic graft-versus-host disease manifesting as polymyositis: an uncommon presentation

Myasthenia gravis and polymyositis as manifestations of chronic graft-versus-host-disease

Rehabilitation following bone marrow transplantation

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