Central and peripheral nervous system immune mediated demyelinating disease after allogeneic hemopoietic stem cell transplantation for hematologic disease Journal Article

  

Authors:	Delios, A. M.; Rosenblum, M.; Jakubowski, A. A.; De Angelis, L. M.
Article Title:	Central and peripheral nervous system immune mediated demyelinating disease after allogeneic hemopoietic stem cell transplantation for hematologic disease
Abstract:	Immune mediated demyelinating disease (IMDD) after allogeneic hemopoietic stem cell transplant (HSCT) is rare and its etiology unclear. In this retrospective study, we identified patients who underwent HSCT between January 1992 and December 2010 and had IMDD post transplant. A total of 1,484 patients received HSCT and 7 (0.5 %) suffered from IMDD; five were men, and the median age was 54 years (range, 29-64 years). HSCT treated acute myeloid leukemia (n = 5), myelodysplastic syndrome (n = 1), and Waldenström macroglobulinemia (n = 1). All received an HLA matched donor graft, related (6), unrelated (1); from the bone marrow (1), peripheral blood stem cell (6); and T-cell depleted, ex vivo (6) or in vivo (1). The median time from transplant to neurologic symptoms was 120 days (range, 60-390 days). Three had acute demyelinating encephalomyelitis (ADEM), three acute inflammatory demyelinating polyradiculopathy (AIDP) and one autonomic neuropathy. Four of six patients tested had hemopoietic mixed chimerism prior to neurologic symptoms and low CD4 + T-cell counts, median 76 (15-500 cells/μL). Two patients had simultaneous systemic graft versus host disease (GVHD). Two patients with ADEM had a spinal cord or brain biopsy which revealed demyelination. No patients had a viral etiology identified in the cerebrospinal fluid. Patients were treated with IV immunoglobulin, high dose steroids and/or rituximab. Five patients had a significant recovery. Response to immune modulators suggests an immune-based etiology. The incidence of de novo autoimmune disease after HSCT for hematological diseases is rare and may be difficult to differentiate from GVHD. © 2012 Springer Science+Business Media, LLC.
Keywords:	adult; clinical article; acute granulocytic leukemia; busulfan; fludarabine; rituximab; drug megadose; bone marrow; peripheral neuropathy; stem cell transplant; cyclophosphamide; immunoglobulin; melphalan; steroid; waldenstroem macroglobulinemia; in vivo study; retrospective study; thiotepa; patient identification; chimera; hla matching; myelodysplastic syndrome; cerebrospinal fluid; peripheral blood stem cell; drug response; graft versus host reaction; allogeneic hematopoietic stem cell transplantation; immunomodulating agent; cd4 lymphocyte count; t cell depletion; autoimmune disease; brain biopsy; ex vivo study; convalescence; neurologic disease; immunopathology; autonomic neuropathy; alemtuzumab; repeated drug dose; hematologic disease; central nervous system disease; demyelination; ephedrine; allogeneic; radiculopathy; encephalomyelitis; demyelinating disease; acute demyelinating encephalomyelitis; acute inflammatory demyelinating polyradiculopathy; demyelinating; pyridostigmine; immune mediated demyelinating disease; spinal cord biopsy
Journal Title:	Journal of Neuro-Oncology
Volume:	110
Issue:	2
ISSN:	0167-594X
Publisher:	Springer
Date Published:	2012-11-01
Start Page:	251
End Page:	256
Language:	English
DOI:	10.1007/s11060-012-0962-9
PROVIDER:	scopus
PUBMED:	22915190
DOI/URL:	http://release-www.scopus.com/inward/record.url?eid=2-s2.0-84867874148&partnerID=40&md5=728cb24c290dcdfe11dfb6221d3a1924
Notes:	--- - "Export Date: 3 December 2012" - "CODEN: JNODD" - "Source: Scopus"

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