Reference : Neuronal surface antibody-mediated encephalopathy as manifestation of chronic graft-v...
Scientific journals : Article
Human health sciences : Hematology
http://hdl.handle.net/2268/228738
Neuronal surface antibody-mediated encephalopathy as manifestation of chronic graft-versus-host disease after allogeneic hematopoietic stem cell transplantation
English
Pirotte, Michèle [Université de Liège - ULiège > > GIGA-R : Hématologie >]
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 mailto [Université de Liège - ULiège > > GIGA-R : Hématologie >]
BEGUIN, Yves mailto [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 >]
2018
Journal of Neuroimmunology
Elsevier B.V.
323
115-118
Yes (verified by ORBi)
International
0165-5728
1872-8421
[en] Allogeneic hematopoietic cell transplant ; Central nervous system ; Graft-versus-host disease ; Neuronal surface antibody-mediated encephalopathy ; PET-CT imaging
[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.
http://hdl.handle.net/2268/228738
10.1016/j.jneuroim.2018.08.003

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