Recurrent Acute Myositis after Allogeneic Bone Marrow Transplantation for Myelodysplasia

Abstract

A 54-year-old woman developed polymyositis 6 months after allogeneic bone marrow transplantation (BMT) for acute myelogenous leukemia transformed from myelodysplasia. At the onset of myositis, the patient had oral dryness, and the histology of oral mucosa was compatible with chronic graft-versus-host disease (GVHD). Muscle biopsy revealed focal muscle necrosis with massive lymphocytic infiltration. She was diagnosed with polymyositis, and the dose of cyclosporine was increased. Three months later, a complete resolution of myositis had been obtained, and the cyclosporine was tapered off. However, 51 months after the first episode of myositis, she again noted severe myalgia and was diagnosed with a recurrence of polymyositis based on high serum creatinine kinase (CK) and the findings of magnetic resonance imaging (MRI). At that time, chronic GVHD in other organs was not present. She achieved a second remission of polymyositis with cyclosporine, and has remained in remission for 4 years. The pathogenesis of myositis can be attributed to the immunologic imbalance characteristic of the post-allogeneic BMT setting.

Fasciitis and myositis: an analysis of muscle-related complications caused by chronic GVHD after allo-SCT

“…7 In myositis, some reported cases showed a recurrence with either a reduction or cessation of immunosuppressant therapy. [10][11][12]20 One of our patients also had recurrent acute myositis after CsA was tapered, and achieved a second remission of polymyositis with CsA. 20 We need to recognize that both fasciitis and myositis caused by chronic GVHD can result in disabilities that reduce the patient's QOL.…”

“…[10][11][12]20 One of our patients also had recurrent acute myositis after CsA was tapered, and achieved a second remission of polymyositis with CsA. 20 We need to recognize that both fasciitis and myositis caused by chronic GVHD can result in disabilities that reduce the patient's QOL. We have to make an early diagnosis and promptly treat fasciitis and myositis to prevent the progression of the complications.…”

“…One patient relapsed after tapering the immunosuppressive treatment, but was successfully treated by increasing the immunosuppression. 20 As of August 2006, all patients are alive. Two patients had residual disabilities, which included muscle atrophy and deformity of vertebrae caused by muscle weakness.…”

Foreign cells in polymyositis: Could stem cell transplantation and pregnancy-derived chimerism lead to the same disease?

“…Thus, PM can be added to the list of potentially allo-autoimmune diseases in which pregnancy-derived microchimerism may play a role. coincidence, rather than as a manifestation of chronic GVHD (Tables 1 and 2) [10][11][12][13][14][15][16][17][18][19][20][21][22][23][24][25][26]. Descriptions of multiple cases of GVHD-PM from single HSCT centers suggested that PM may be part of GVHD [1•,27,28].…”

Hemosiderin deposits in chronic graft‐vs.‐host disease related myopathy

“…cGVHD‐associated polymyositis or myopathy usually occurs 2–5 yr after SCT and is typically pronounced in the proximal lower limb (4, 6, 11–13). The majority of the patients show cGVHD‐involvement of organ systems other than the skeletal muscles (6, 14–16), but it seems that polymyositis or myopathy may occur as a unique manifestation of cGVHD (10, 17).…”