Severe allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation

Abstract Allogeneic hematopoietic stem cell transplantation (alloHSCT) is a curative treatment for hematologic malignancies. Acute and chronic graft-versus-host disease (GvHD) are the major immune-mediated complications after alloHSCT. However, there is controversy whether neurologic complications a...

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Autores principales:	Martin W. Hümmert, Michael Stadler, Lothar Hambach, Stefan Gingele, Martin Bredt, Mike P. Wattjes, Gudrun Göhring, Letizia Venturini, Nora Möhn, Martin Stangel, Corinna Trebst, Arnold Ganser, Florian Wegner, Thomas Skripuletz
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2021
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/c675c306039d4e41a2256268f7fe09c0
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spelling	oai:doaj.org-article:c675c306039d4e41a2256268f7fe09c02021-12-02T18:27:48ZSevere allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation10.1038/s41598-021-87989-z2045-2322https://doaj.org/article/c675c306039d4e41a2256268f7fe09c02021-04-01T00:00:00Zhttps://doi.org/10.1038/s41598-021-87989-zhttps://doaj.org/toc/2045-2322Abstract Allogeneic hematopoietic stem cell transplantation (alloHSCT) is a curative treatment for hematologic malignancies. Acute and chronic graft-versus-host disease (GvHD) are the major immune-mediated complications after alloHSCT. However, there is controversy whether neurologic complications after alloHSCT might represent manifestations of GvHD. We report three patients who acquired distinct, severe immune-mediated peripheral or central nervous system diseases after alloHSCT without other, concomitant GvHD manifestations. One patient had been diagnosed with B-cell chronic lymphocytic leukemia and two patients with high risk myelodysplastic syndrome. Patient #1 presented as LGI1- and GAD-IgG positive immune-mediated encephalitis, patient #2 was diagnosed with MOG-IgG positive encephalomyelitis, and patient #3 had chronic inflammatory polyneuropathy associated with SSA(Ro)-IgG positive Sjögren’s syndrome. 100% donor chimerism was detectable in the peripheral blood in all three. The specific antibodies were undetectable in donors’ and patients’ blood before alloHSCT suggesting that the antibodies had arisen from the transplanted donor immune system. Early intensive immunotherapy led to improvement of clinical symptoms and stability of the neurological disease, however, at the cost of losing the graft-versus-malignancy effect in one patient. In conclusion, we provide evidence of isolated, severe allo-immune diseases of the peripheral and central nervous system as complications of alloHSCT (“neuro-GvHD”). Interdisciplinary surveillance and thorough diagnostic work-up are needed for early diagnosis and treatment of neuro-immunologic complications after alloHSCT to improve the otherwise poor outcome.Martin W. HümmertMichael StadlerLothar HambachStefan GingeleMartin BredtMike P. WattjesGudrun GöhringLetizia VenturiniNora MöhnMartin StangelCorinna TrebstArnold GanserFlorian WegnerThomas SkripuletzNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 11, Iss 1, Pp 1-8 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Martin W. Hümmert Michael Stadler Lothar Hambach Stefan Gingele Martin Bredt Mike P. Wattjes Gudrun Göhring Letizia Venturini Nora Möhn Martin Stangel Corinna Trebst Arnold Ganser Florian Wegner Thomas Skripuletz Severe allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation
description	Abstract Allogeneic hematopoietic stem cell transplantation (alloHSCT) is a curative treatment for hematologic malignancies. Acute and chronic graft-versus-host disease (GvHD) are the major immune-mediated complications after alloHSCT. However, there is controversy whether neurologic complications after alloHSCT might represent manifestations of GvHD. We report three patients who acquired distinct, severe immune-mediated peripheral or central nervous system diseases after alloHSCT without other, concomitant GvHD manifestations. One patient had been diagnosed with B-cell chronic lymphocytic leukemia and two patients with high risk myelodysplastic syndrome. Patient #1 presented as LGI1- and GAD-IgG positive immune-mediated encephalitis, patient #2 was diagnosed with MOG-IgG positive encephalomyelitis, and patient #3 had chronic inflammatory polyneuropathy associated with SSA(Ro)-IgG positive Sjögren’s syndrome. 100% donor chimerism was detectable in the peripheral blood in all three. The specific antibodies were undetectable in donors’ and patients’ blood before alloHSCT suggesting that the antibodies had arisen from the transplanted donor immune system. Early intensive immunotherapy led to improvement of clinical symptoms and stability of the neurological disease, however, at the cost of losing the graft-versus-malignancy effect in one patient. In conclusion, we provide evidence of isolated, severe allo-immune diseases of the peripheral and central nervous system as complications of alloHSCT (“neuro-GvHD”). Interdisciplinary surveillance and thorough diagnostic work-up are needed for early diagnosis and treatment of neuro-immunologic complications after alloHSCT to improve the otherwise poor outcome.
format	article
author	Martin W. Hümmert Michael Stadler Lothar Hambach Stefan Gingele Martin Bredt Mike P. Wattjes Gudrun Göhring Letizia Venturini Nora Möhn Martin Stangel Corinna Trebst Arnold Ganser Florian Wegner Thomas Skripuletz
author_facet	Martin W. Hümmert Michael Stadler Lothar Hambach Stefan Gingele Martin Bredt Mike P. Wattjes Gudrun Göhring Letizia Venturini Nora Möhn Martin Stangel Corinna Trebst Arnold Ganser Florian Wegner Thomas Skripuletz
author_sort	Martin W. Hümmert
title	Severe allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation
title_short	Severe allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation
title_full	Severe allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation
title_fullStr	Severe allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation
title_full_unstemmed	Severe allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation
title_sort	severe allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation
publisher	Nature Portfolio
publishDate	2021
url	https://doaj.org/article/c675c306039d4e41a2256268f7fe09c0
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Severe allo-immune antibody-associated peripheral and central nervous system diseases after allogeneic hematopoietic stem cell transplantation

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