Cargando…

Prion pathogenesis is unaltered in the absence of SIRPα-mediated "don't-eat-me" signaling

Prion diseases are neurodegenerative conditions caused by misfolding of the prion protein, leading to conspicuous neuronal loss and intense microgliosis. Recent experimental evidence point towards a protective role of microglia against prion-induced neurodegeneration, possibly through elimination of...

Descripción completa

Detalles Bibliográficos
Autores principales: Nuvolone, Mario, Paolucci, Marta, Sorce, Silvia, Kana, Veronika, Moos, Rita, Matozaki, Takashi, Aguzzi, Adriano
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Public Library of Science 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5435345/
https://www.ncbi.nlm.nih.gov/pubmed/28545141
http://dx.doi.org/10.1371/journal.pone.0177876
_version_ 1783237219688906752
author Nuvolone, Mario
Paolucci, Marta
Sorce, Silvia
Kana, Veronika
Moos, Rita
Matozaki, Takashi
Aguzzi, Adriano
author_facet Nuvolone, Mario
Paolucci, Marta
Sorce, Silvia
Kana, Veronika
Moos, Rita
Matozaki, Takashi
Aguzzi, Adriano
author_sort Nuvolone, Mario
collection PubMed
description Prion diseases are neurodegenerative conditions caused by misfolding of the prion protein, leading to conspicuous neuronal loss and intense microgliosis. Recent experimental evidence point towards a protective role of microglia against prion-induced neurodegeneration, possibly through elimination of prion-containing apoptotic bodies. The molecular mechanisms by which microglia recognize and eliminate apoptotic cells in the context of prion diseases are poorly defined. Here we investigated the possible involvement of signal regulatory protein α (SIRPα), a key modulator of host cell phagocytosis; SIRPα is encoded by the Sirpa gene that is genetically linked to the prion gene Prnp. We found that Sirpa transcripts are highly enriched in microglia cells within the brain. However, Sirpa mRNA levels were essentially unaltered during the course of experimental prion disease despite upregulation of other microglia-enriched transcripts. To study the involvement of SIRPα in prion pathogenesis in vivo, mice expressing a truncated SIRPα protein unable to inhibit phagocytosis were inoculated with rodent-adapted scrapie prions of the 22L strain. Homozygous and heterozygous Sirpa mutants and wild-type mice experienced similar incubation times after inoculation with either of two doses of 22L prions. Moreover, the extent of neuronal loss, microgliosis and abnormal prion protein accumulation was not significantly affected by Sirpa genotypes. Collectively, these data indicate that SIRPα-mediated phagocytosis is not a major determinant in prion disease pathogenesis. It will be important to search for additional candidates mediating prion phagocytosis, as this mechanism may represent an important target of antiprion therapies.
format Online
Article
Text
id pubmed-5435345
institution National Center for Biotechnology Information
language English
publishDate 2017
publisher Public Library of Science
record_format MEDLINE/PubMed
spelling pubmed-54353452017-05-26 Prion pathogenesis is unaltered in the absence of SIRPα-mediated "don't-eat-me" signaling Nuvolone, Mario Paolucci, Marta Sorce, Silvia Kana, Veronika Moos, Rita Matozaki, Takashi Aguzzi, Adriano PLoS One Research Article Prion diseases are neurodegenerative conditions caused by misfolding of the prion protein, leading to conspicuous neuronal loss and intense microgliosis. Recent experimental evidence point towards a protective role of microglia against prion-induced neurodegeneration, possibly through elimination of prion-containing apoptotic bodies. The molecular mechanisms by which microglia recognize and eliminate apoptotic cells in the context of prion diseases are poorly defined. Here we investigated the possible involvement of signal regulatory protein α (SIRPα), a key modulator of host cell phagocytosis; SIRPα is encoded by the Sirpa gene that is genetically linked to the prion gene Prnp. We found that Sirpa transcripts are highly enriched in microglia cells within the brain. However, Sirpa mRNA levels were essentially unaltered during the course of experimental prion disease despite upregulation of other microglia-enriched transcripts. To study the involvement of SIRPα in prion pathogenesis in vivo, mice expressing a truncated SIRPα protein unable to inhibit phagocytosis were inoculated with rodent-adapted scrapie prions of the 22L strain. Homozygous and heterozygous Sirpa mutants and wild-type mice experienced similar incubation times after inoculation with either of two doses of 22L prions. Moreover, the extent of neuronal loss, microgliosis and abnormal prion protein accumulation was not significantly affected by Sirpa genotypes. Collectively, these data indicate that SIRPα-mediated phagocytosis is not a major determinant in prion disease pathogenesis. It will be important to search for additional candidates mediating prion phagocytosis, as this mechanism may represent an important target of antiprion therapies. Public Library of Science 2017-05-17 /pmc/articles/PMC5435345/ /pubmed/28545141 http://dx.doi.org/10.1371/journal.pone.0177876 Text en © 2017 Nuvolone et al http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Research Article
Nuvolone, Mario
Paolucci, Marta
Sorce, Silvia
Kana, Veronika
Moos, Rita
Matozaki, Takashi
Aguzzi, Adriano
Prion pathogenesis is unaltered in the absence of SIRPα-mediated "don't-eat-me" signaling
title Prion pathogenesis is unaltered in the absence of SIRPα-mediated "don't-eat-me" signaling
title_full Prion pathogenesis is unaltered in the absence of SIRPα-mediated "don't-eat-me" signaling
title_fullStr Prion pathogenesis is unaltered in the absence of SIRPα-mediated "don't-eat-me" signaling
title_full_unstemmed Prion pathogenesis is unaltered in the absence of SIRPα-mediated "don't-eat-me" signaling
title_short Prion pathogenesis is unaltered in the absence of SIRPα-mediated "don't-eat-me" signaling
title_sort prion pathogenesis is unaltered in the absence of sirpα-mediated "don't-eat-me" signaling
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5435345/
https://www.ncbi.nlm.nih.gov/pubmed/28545141
http://dx.doi.org/10.1371/journal.pone.0177876
work_keys_str_mv AT nuvolonemario prionpathogenesisisunalteredintheabsenceofsirpamediateddonteatmesignaling
AT paoluccimarta prionpathogenesisisunalteredintheabsenceofsirpamediateddonteatmesignaling
AT sorcesilvia prionpathogenesisisunalteredintheabsenceofsirpamediateddonteatmesignaling
AT kanaveronika prionpathogenesisisunalteredintheabsenceofsirpamediateddonteatmesignaling
AT moosrita prionpathogenesisisunalteredintheabsenceofsirpamediateddonteatmesignaling
AT matozakitakashi prionpathogenesisisunalteredintheabsenceofsirpamediateddonteatmesignaling
AT aguzziadriano prionpathogenesisisunalteredintheabsenceofsirpamediateddonteatmesignaling