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Cystatin F (Cst7) drives sex-dependent changes in microglia in an amyloid-driven model of Alzheimer's disease.
Daniels, Michael J D; Lefevre, Lucas; Szymkowiak, Stefan; Drake, Alice; McCulloch, Laura; Tzioras, Makis; Barrington, Jack; Dando, Owen R; He, Xin; Mohammad, Mehreen; Sasaguri, Hiroki; Saito, Takashi; Saido, Takaomi C; Spires-Jones, Tara L; McColl, Barry W.
Afiliación
  • Daniels MJD; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • Lefevre L; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • Szymkowiak S; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • Drake A; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • McCulloch L; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • Tzioras M; Centre for Inflammation Research, Institute for Regeneration and Repair, The University of Edinburgh, Edinburgh, United Kingdom.
  • Barrington J; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • Dando OR; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • He X; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • Mohammad M; Simons Initiative for the Developing Brain, University of Edinburgh, Edinburgh, United Kingdom.
  • Sasaguri H; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • Saito T; Simons Initiative for the Developing Brain, University of Edinburgh, Edinburgh, United Kingdom.
  • Saido TC; UK Dementia Research Insitute at The University of Edinburgh, Edinburgh, United Kingdom.
  • Spires-Jones TL; Department of Neurology and Neurological Science, Graduate School of Medicine, Tokyo Medical and Dental University, Tokyo, Japan.
  • McColl BW; Laboratory for Proteolytic Neuroscience, RIKEN Brain Science Institute, Wako, Japan.
Elife ; 122023 12 12.
Article en En | MEDLINE | ID: mdl-38085657
Microglial endolysosomal (dys)function is strongly implicated in neurodegenerative disease. Transcriptomic studies show that a microglial state characterised by a set of genes involved in endolysosomal function is induced in both mouse Alzheimer's disease (AD) models and human AD brain, and that the emergence of this state is emphasised in females. Cst7 (encoding cystatin F) is among the most highly upregulated genes in these microglia. However, despite such striking and robust upregulation, the function of Cst7 in neurodegenerative disease is not understood. Here, we crossed Cst7-/- mice with the AppNL-G-F mouse to test the role of Cst7 in a model of amyloid-driven AD. Surprisingly, we found that Cst7 plays a sexually dimorphic role regulating microglia in this model. In females, Cst7-/-AppNL-G-F microglia had greater endolysosomal gene expression, lysosomal burden, and amyloid beta (Aß) burden in vivo and were more phagocytic in vitro. However, in males, Cst7-/-AppNL-G-F microglia were less inflammatory and had a reduction in lysosomal burden but had no change in Aß burden. Overall, our study reveals functional roles for one of the most commonly upregulated genes in microglia across disease models, and the sex-specific profiles of Cst7-/--altered microglial disease phenotypes. More broadly, the findings raise important implications for AD including crucial questions on sexual dimorphism in neurodegenerative disease and the interplay between endolysosomal and inflammatory pathways in AD pathology.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Cistatinas / Enfermedades Neurodegenerativas / Enfermedad de Alzheimer Límite: Animals / Female / Humans / Male Idioma: En Revista: Elife Año: 2023 Tipo del documento: Article País de afiliación: Reino Unido Pais de publicación: Reino Unido

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Cistatinas / Enfermedades Neurodegenerativas / Enfermedad de Alzheimer Límite: Animals / Female / Humans / Male Idioma: En Revista: Elife Año: 2023 Tipo del documento: Article País de afiliación: Reino Unido Pais de publicación: Reino Unido