Neurologic Complications of Graft-Versus-Host Disease

Nelson, Kevin R.; McQuillen, Michael P.

doi:10.1016/s0733-8619(18)30877-6

Cited by 36 publications

(29 citation statements)

References 67 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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“…6,7 The mean time from allogeneic BMT to onset of myositis ranges from 100 days to 54 months. 6,8 As demonstrated in the case we describe, patients with GVHD-related polymyositis usually have other manifestations of acute or chronic GVHD. Polymyositis has been reported as the sole manifestation of chronic GVHD.…”

Section: Discussionmentioning

confidence: 69%

“…Patients usually require treatment for 5-30 months and the overall prognosis appears to depend more on the activity of the chronic GVHD rather than the severity of the polymyositis. 8 Although a muscle biopsy was not obtained, due to concerns about wound healing in the setting of extensive skin GVHD and steroid use, we believe that there is sufficient evidence to support a diagnosis of GVHD-related polymyositis in this case. The clinical features of polymyositis with elevated muscle enzymes, along with evidence of inflammation on MRI, all occurred in the setting of biopsy proven chronic GVHD.…”

Section: Discussionmentioning

confidence: 73%

“…7 GVHD-related polymyositis usually responds to corticosteroids but may relapse as the corticosteroids are tapered. 8 In the case we report additional immunosuppressive therapy was required to manage the severe GVHD-related polymyositis. Patients usually require treatment for 5-30 months and the overall prognosis appears to depend more on the activity of the chronic GVHD rather than the severity of the polymyositis.…”

Section: Discussionmentioning

confidence: 89%

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Chronic graft-versus-host disease-related polymyositis as a cause of respiratory failure following allogeneic bone marrow transplant

Leano

Miller

White

2000

Bone Marrow Transplant

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Summary:An unusual case of respiratory failure and dropped head syndrome as a complication of severe chronic graft-versus-host disease (GVHD)-related polymyositis is described. The patient required tracheostomy and mechanical ventilation but recovered following treatment with aggressive immunosuppression and intensive rehabilitation. The differential diagnoses of muscle weakness in the bone marrow transplant (BMT) patient and the dropped head syndrome are both discussed. To our knowledge, this is the first reported case of respiratory failure requiring mechanical ventilation occurring as a complication of GVHD-related polymyositis. Bone Marrow Transplantation (2000) 26, 1117-1120. Keywords: polymyositis; GVHD; respiratory failure; BMT; dropped head syndrome Respiratory failure in the setting of bone marrow transplantation (BMT) is usually acute in onset and occurs due to a number of well described but poorly understood clinical syndromes that occur in this patient population. The clinical syndromes include diffuse alveolar hemorrhage, 1 the idiopathic pneumonia syndrome, 2 septic shock, 3 pneumonia due to conventional and opportunistic organisms 4 or bronchiolitis obliterans.5 Chronic respiratory failure as a complication of muscle weakness in this patient population is not well described and appears to be very uncommon. Polymyositis is a recognized but unusual complication of chronic graft-versus-host disease (GVHD) due to BMT. 6 We report a patient who developed GVHD-related polymyositis that resulted in profound weakness of both the extensor muscles of the neck and muscles of respiration. The patient gradually developed respiratory failure and ultimately required a tracheostomy and mechanical ventilation. To our knowledge, GVHD as a cause of chronic respiratory failure has not been previously described. The differential diagnoses of respiratory muscle weakness in the BMT patient and the 'dropped head syndrome' are both discussed. Case reportA 37-year-old female who had undergone a matched unrelated allogeneic BMT 2 years prior to admission presented with a several month history of progressive weakness of the muscles of both shoulders, neck and thighs. She had difficulty performing simple tasks such as combing her hair and brushing her teeth. In addition, she had diffuse myalgias and dysphagia for both liquids and solids. Ten years previously she had been diagnosed with acute myeloid leukemia (M2) (AML) that responded to chemotherapy. The disease relapsed 7 years later and she was treated with induction chemotherapy, two cycles of consolidation followed by a matched (6/6) unrelated allogeneic BMT from an ABO incompatible donor. She received a standard conditioning regimen comprising cyclophosphamide and total body irradiation. Routine pulmonary function tests performed prior to BMT were all normal. Her course following BMT was complicated by grade 1 acute and then extensive chronic GVHD, involving primarily the oral mucosa and skin. This was treated with corticosteroids (prednisone 10-20 mg per day) and cyclos...

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Development of Lhermitte's sign after bone marrow transplantation

Wen¹,

Blanchard²,

Block³

et al. 1992

Cancer

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The authors observed Lhermitte's sign in four patients after bone marrow transplantation (BMT) for hematologic malignancies. Three patients had acute myelogenous leukemia (AML), and one had chronic myelogenous leukemia. Before BMT, the patients with AML received daunorubicin, cytosine arabinoside and etoposide, whereas the patient with chronic myelogenous leukemia received hydroxyurea. One patient with AML received MY‐9 antibody‐depleted autologous BMT. The other patients received human lymphocyte antigen‐identical, allogeneic BMT. Preparative therapy for BMT was cytosine arabinoside, cyclophosphamide, and total body exposure to radiation for two patients, and busulfan, cyclophosphamide, and no exposure to radiation in two other patients. Lhermitte's sign appeared 4 to 8 months after BMT and resolved spontaneously after 2 to 5 months. Neurologic sequelae had developed in none of the patients 16 to 34 months after BMT. No unifying etiologic factor could be identified in these patients. The development of Lhermitte's sign after BMT appears to be a benign, self‐limited phenomenon that requires no specific treatment.

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Neurologic Complications of Graft-Versus-Host Disease

Cited by 36 publications

References 67 publications

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

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

Chronic graft-versus-host disease-related polymyositis as a cause of respiratory failure following allogeneic bone marrow transplant

Development of Lhermitte's sign after bone marrow transplantation

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