Article (Scientific journals)
Neuronal surface antibody-mediated encephalopathy as manifestation of chronic graft-versus-host disease after allogeneic hematopoietic stem cell transplantation
Pirotte, Michelle; Forte, Florence; Lutteri, Laurence et al.
2018In Journal of Neuroimmunology, 323, p. 115-118
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Keywords :
Allogeneic hematopoietic cell transplant; Central nervous system; Graft-versus-host disease; Neuronal surface antibody-mediated encephalopathy; PET-CT imaging
Abstract :
[en] Although it remained controversial for a long time, central nervous system (CNS) involvement of graft-versus-host disease (GVHD) is now becoming recognized as a real nosological entity. Previous case reports have suggested heterogeneous clinical presentations and it is not excluded that the whole spectrum of manifestations has not yet been fully described. Here, we report the case of a 58-year-old man with chronic GVHD who developed a rapidly ingravescent encephalopathy. There was no evidence for CNS immune-mediated lesions on conventional imaging nor for cellular infiltration in the cerebrospinal fluid. Serum analyses revealed the presence of anti-neuronal antibodies directed against anti-contactin-associated protein 2 (anti-Caspr2), a protein associated with voltage-gated potassium neuronal channels. Functional imaging with 2-deoxy-2-[fluorine-18] fluoro- D-glucose integrated with computed tomography (18F-FDG PET-CT) demonstrated diffuse cortical and subcortical hypometabolism. The patient was treated with a combination of immunosuppressive agents (corticosteroids, cyclophosphamide and rituximab) and progressively recovered normal neurocognitive functions. Taken together, these data suggest that CNS-GVHD may manifest as a reversible antibody-mediated functional encephalopathy. This report suggests for the first time the interest of screening for anti-neuronal antibodies and functional imaging with brain 18F-FDG PET-CT in diagnosing this severe complication of allogeneic hematopoietic cell transplantation (alloHSCT). © 2018 Elsevier B.V.
Disciplines :
Hematology
Author, co-author :
Pirotte, Michelle  ;  Université de Liège - ULiège > GIGA - MASSGPFS
Forte, Florence  ;  Department of Neurology, CHU of Liège and ULiege, CHU Sart-Tilman, Liège, Belgium
Lutteri, Laurence ;  Université de Liège - ULiège > Département de pharmacie > Chimie médicale
WILLEMS, Evelyne ;  Centre Hospitalier Universitaire de Liège - CHU > Département de médecine interne > Service d'hématologie clinique
DURAN, Unal ;  Centre Hospitalier Universitaire de Liège - CHU > Département de Physique Médicale > Service médical de radiodiagnostic
Belle, Ludovic ;  Université de Liège - ULiège > GIGA-R : Hématologie
Baron, Frédéric  ;  Université de Liège - ULiège > GIGA-R : Hématologie
BEGUIN, Yves  ;  Centre Hospitalier Universitaire de Liège - CHU > Département de médecine interne > Service d'hématologie clinique
Maquet, Pierre  ;  Université de Liège - ULiège > Département des sciences cliniques > Neurologie
BODART, Olivier   ;  Centre Hospitalier Universitaire de Liège - CHU > Département de médecine interne > Service de neurologie
Servais, Sophie  ;  Université de Liège - ULiège > GIGA-R : Hématologie
 These authors have contributed equally to this work.
Language :
English
Title :
Neuronal surface antibody-mediated encephalopathy as manifestation of chronic graft-versus-host disease after allogeneic hematopoietic stem cell transplantation
Publication date :
2018
Journal title :
Journal of Neuroimmunology
ISSN :
0165-5728
eISSN :
1872-8421
Publisher :
Elsevier B.V.
Volume :
323
Pages :
115-118
Peer reviewed :
Peer Reviewed verified by ORBi
Available on ORBi :
since 19 October 2018

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