Encephalomyelitis Mimicking Multiple Sclerosis Associated with Chronic Graft-Versus-Host Disease after Allogeneic Bone Marrow Transplantation

Matsuo, Yoko; Kamezaki, Kenjiro; Takeishi, Shoichiro; Takenaka, Katsuto; Eto, Tetsuya; Nonami, Atsushi; Miyamoto, Toshihiro; Iwasaki, Hiromi; Harada, Naoki; Nagafuji, Koji; Teshima, Takanori; Akashi, Koichi

doi:10.2169/internalmedicine.48.2003

Cited by 42 publications

(29 citation statements)

References 13 publications

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“…A case reported by Matsuo et al 9 described a patient with a relapsing inflammatory illness of the nervous system that began with a unilateral optic neuritis before involving the other optic nerve, the brain, and spinal cord, and stabilised with steroids and cyclosporin A. Openshaw et al 10 described two patients with myelopathy, one of whom developed an optic neuropathy during steroid withdrawal.…”

Section: Discussionmentioning

confidence: 99%

Bilateral Inflammatory Optic Neuropathy Related to Graft versus Host Disease Following Allogeneic Bone Marrow Transplantation for Hodgkin Disease

Moesen

Kidd

2014

Neuro-Ophthalmology

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Graft versus host disease (GvHD) is a common and often troublesome complication of allogeneic bone marrow transplantation. Neurological complications usually involve the peripheral nervous system and muscle, but the central nervous system may be affected. When an optic neuropathy develops, it is often difficult to determine the cause quickly; infective complications and drug toxicity may have arisen, but an inflammatory disorder due to GvHD should also be considered, particularly since treatment with steroids and immune suppression may improve the outcome significantly. This brief case report shows how this may be the case and reviews our current understanding of the pathophysiology and treatment of the disorder within the nervous system.

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Section: Discussionmentioning

confidence: 99%

Bilateral Inflammatory Optic Neuropathy Related to Graft versus Host Disease Following Allogeneic Bone Marrow Transplantation for Hodgkin Disease

Moesen

Kidd

2014

Neuro-Ophthalmology

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“…The pathophysiology of CNS-GvHD is pleomorphic, and underlying pathological processes include cerebrovascular disease,13 14 demyelinating disease of the CNS15 and immune-mediated encephalitis 6 13–20. Patients with biopsy-proven CNS-GvHD6 present with a variety of clinical symptoms including, seizures,3 confusion, progressive spasticity,21 irritability, proximal muscular lower extremity weakness,6 dysphagia, dysarthria, hemianopsia, aphasia, paresis,14 progressive clouding of consciousness and coma 22.…”

Section: Discussionmentioning

confidence: 99%

Central nervous system graft-versus-host disease (CNS-GvHD) after allogeneic haematopoietic stem cell transplantation

et al. 2018

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A 60-year-old man presented with impaired consciousness and psychomotor agitation after a second allogeneic haematopoietic stem cell transplantation (HSCT) from a matched unrelated donor for acute myeloid leukaemia. Clinical, biological and radiological evidence suggested a diagnosis of central nervous system graft-versus-host disease (CNS-GvHD). After intrathecal infusion of methylprednisolone, the clinical symptoms as well as the radiological abnormalities disappeared. The present report illustrates the difficulties in the diagnosis and the management of CNS-GvHD, a very rare and still challenging neurological complication that can occur after allogeneic HSCT.

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“…3 However, the CNS is remarkably spared in cases of graft vs host disease seen following BMT, 4 and immune-mediated complications are exceptional. 5,6 This, and the variable susceptibility of individuals to tissuespecific complications, is unexplained. Identification of MS risk haplotypes here may be relevant since they confer significant risk and likely localize where environment may interact.…”

Section: Discussionmentioning

confidence: 99%

De Novo Relapsing-Remitting Multiple Sclerosis Following Autologous Stem Cell Transplantation

Armstrong

Elston²,

Hatton³

et al. 2010

Neurology

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We report a 62-year-old man with a relapsingremitting phenotype characteristic of early adulthood multiple sclerosis (MS). Onset with sequential, but rapidly remitting episodes of optic neuritis, transverse myelitis, and a brainstem syndrome followed autologous stem cell transplant (SCT) for plasma cell leukemia. This occurrence in a compound heterozygote for 2 MS-associated class II major histocompatibility haplotypes further implicates immune dysregulation in the etiopathogenesis of MS.Case report. A 62-year-old northern European man, without previous neurologic dysfunction, presented with weight loss, malaise, and thoracic back pain. Investigations revealed plasma cells in the peripheral blood, an immunoglobulin G (IgG) paraprotein of 50.7 g/L Ϫ1 with associated immunoparesis, and ␤ 2 microglobulin of 85.9 mg/ L Ϫ1 . Serum calcium was 3.1 mmol and there were lytic lesions and associated collapse of multiple thoracic vertebrae. Stage III plasma cell leukemia was diagnosed. He underwent 5 cycles of cytarabine/thalidomide/danarubicin chemotherapy and, 5 months after diagnosis, an autologous SCT with reinfusion of CD34ϩ stem cells following marrow ablation with melphalan. Within 6 months, he was in remission with an undetectable paraprotein and normal blood indices.Eight months following SCT, he developed isolated right retroorbital pain with central visual loss reaching a nadir at 1 week. A relative afferent papillary defect, an acuity of 6/9 (6/4.4 left), reduced color vision (Ishihara 6/17), and neurophysiologic data supported a clinical diagnosis of optic neuritis (figure, A and B). No significant white matter abnormalities were evident outside the affected optic nerve (figure, C). A full recovery ensued without treatment.Nine months following SCT, he developed a band of burning midthoracic discomfort that evolved over 1 week to include dysesthesia and allodynia below the level. Two weeks later, symptoms had fully resolved.Ten months after SCT, his gait became unsteady and his hands clumsy over 4 days followed by vertigo, diplopia, and oscillopsia. He became unable to mobilize safely. Examination revealed limited abduction of the left eye, degraded horizontal pursuit and hypometric saccades, gait ataxia, and decomposition of left arm movement. MRI showed new high T2 signal lesions in the left pons and cerebellar peduncle (figure, D). Blood indices were normal and no paraprotein detectable. CSF was acellular with a normal IgG:albumin ratio (0.12) and no oligoclonal bands detectable by immunofixation. Prior exposure to Epstein-Barr virus was indicated by the detection of IgG against Epstein-Barr virus-capsid antigen preand post-SCT. No treatment was given. By day 5, he mobilized with assistance, and in 2 weeks he had recovered. Tissue typing revealed he was a HLA-DRB1*15/HLA-DRB1*17 heterozygote. Discussion.Remitting episodes of optic neuritis, probable transverse myelitis, and a brainstem syndrome occurring sequentially over 4 months allow the diagnosis of relapsing-remitting MS on clinical criteria. The absenc...

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Encephalomyelitis Mimicking Multiple Sclerosis Associated with Chronic Graft-Versus-Host Disease after Allogeneic Bone Marrow Transplantation

Abstract: We describe a case of encephalomyelitis mimicking multiple sclerosis associated with chronic graft-versushost disease (GVHD)

Cited by 42 publications

References 13 publications

Bilateral Inflammatory Optic Neuropathy Related to Graft versus Host Disease Following Allogeneic Bone Marrow Transplantation for Hodgkin Disease

Bilateral Inflammatory Optic Neuropathy Related to Graft versus Host Disease Following Allogeneic Bone Marrow Transplantation for Hodgkin Disease

Central nervous system graft-versus-host disease (CNS-GvHD) after allogeneic haematopoietic stem cell transplantation

De Novo Relapsing-Remitting Multiple Sclerosis Following Autologous Stem Cell Transplantation

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