Structural and functional studies of the VAPB-PTPIP51 ER-mitochondria tethering proteins in neurodegenerative diseases

Abstract Signaling between the endoplasmic reticulum (ER) and mitochondria regulates many of the seemingly disparate physiological functions that are damaged in neurodegenerative diseases such as Alzheimer’s disease, Parkinson’s disease, frontotemporal dementia (FTD) and amyotrophic lateral sclerosi...

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Main Authors: Kerry Blair, Raquel Martinez-Serra, Philippe Gosset, Sandra M. Martín-Guerrero, Gábor M. Mórotz, Joseph Atherton, Jacqueline C. Mitchell, Andrea Markovinovic, Christopher C. J. Miller
Format: Article
Language:English
Published: BMC 2025-03-01
Series:Acta Neuropathologica Communications
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Online Access:https://doi.org/10.1186/s40478-025-01964-7
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author Kerry Blair
Raquel Martinez-Serra
Philippe Gosset
Sandra M. Martín-Guerrero
Gábor M. Mórotz
Joseph Atherton
Jacqueline C. Mitchell
Andrea Markovinovic
Christopher C. J. Miller
author_facet Kerry Blair
Raquel Martinez-Serra
Philippe Gosset
Sandra M. Martín-Guerrero
Gábor M. Mórotz
Joseph Atherton
Jacqueline C. Mitchell
Andrea Markovinovic
Christopher C. J. Miller
author_sort Kerry Blair
collection DOAJ
description Abstract Signaling between the endoplasmic reticulum (ER) and mitochondria regulates many of the seemingly disparate physiological functions that are damaged in neurodegenerative diseases such as Alzheimer’s disease, Parkinson’s disease, frontotemporal dementia (FTD) and amyotrophic lateral sclerosis (ALS). A number of studies have now demonstrated that ER-mitochondria signaling is perturbed in these diseases and there is evidence that this may be a driving mechanism in disease onset and progression. VAPB and PTPIP51 are ER-mitochondria tethering proteins; VAPB is an ER protein and PTPIP51 is an outer mitochondrial membrane protein and the two proteins interact to enable inter-organelle signaling. The VAPB-PTPIP51 interaction is disrupted in Alzheimer’s disease, Parkinson’s disease, FTD and ALS. Here we review the roles of VAPB and PTPIP51 in ER-mitochondria signaling and the mechanisms by which neurodegenerative disease insults may disrupt the VAPB-PTPIP51 interaction.
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institution Kabale University
issn 2051-5960
language English
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publisher BMC
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series Acta Neuropathologica Communications
spelling doaj-art-8d2fa117b9cf4039ad17be12c36bcefb2025-08-20T03:45:41ZengBMCActa Neuropathologica Communications2051-59602025-03-011311910.1186/s40478-025-01964-7Structural and functional studies of the VAPB-PTPIP51 ER-mitochondria tethering proteins in neurodegenerative diseasesKerry Blair0Raquel Martinez-Serra1Philippe Gosset2Sandra M. Martín-Guerrero3Gábor M. Mórotz4Joseph Atherton5Jacqueline C. Mitchell6Andrea Markovinovic7Christopher C. J. Miller8Department of Basic and Clinical Neuroscience, Institute of Psychiatry, Psychology and Neuroscience, King’s College LondonDepartment of Basic and Clinical Neuroscience, Institute of Psychiatry, Psychology and Neuroscience, King’s College LondonDepartment of Basic and Clinical Neuroscience, Institute of Psychiatry, Psychology and Neuroscience, King’s College LondonDepartment of Basic and Clinical Neuroscience, Institute of Psychiatry, Psychology and Neuroscience, King’s College LondonDepartment of Basic and Clinical Neuroscience, Institute of Psychiatry, Psychology and Neuroscience, King’s College LondonRandall Centre for Cell and Molecular Biophysics, School of Basic and Medical Biosciences, King’s College LondonDepartment of Basic and Clinical Neuroscience, Institute of Psychiatry, Psychology and Neuroscience, King’s College LondonDepartment of Basic and Clinical Neuroscience, Institute of Psychiatry, Psychology and Neuroscience, King’s College LondonDepartment of Basic and Clinical Neuroscience, Institute of Psychiatry, Psychology and Neuroscience, King’s College LondonAbstract Signaling between the endoplasmic reticulum (ER) and mitochondria regulates many of the seemingly disparate physiological functions that are damaged in neurodegenerative diseases such as Alzheimer’s disease, Parkinson’s disease, frontotemporal dementia (FTD) and amyotrophic lateral sclerosis (ALS). A number of studies have now demonstrated that ER-mitochondria signaling is perturbed in these diseases and there is evidence that this may be a driving mechanism in disease onset and progression. VAPB and PTPIP51 are ER-mitochondria tethering proteins; VAPB is an ER protein and PTPIP51 is an outer mitochondrial membrane protein and the two proteins interact to enable inter-organelle signaling. The VAPB-PTPIP51 interaction is disrupted in Alzheimer’s disease, Parkinson’s disease, FTD and ALS. Here we review the roles of VAPB and PTPIP51 in ER-mitochondria signaling and the mechanisms by which neurodegenerative disease insults may disrupt the VAPB-PTPIP51 interaction.https://doi.org/10.1186/s40478-025-01964-7Neurodegenerative diseasesFrontotemporal dementiaAmyotrophic lateral sclerosisAlzheimer’s diseaseParkinson’s disease
spellingShingle Kerry Blair
Raquel Martinez-Serra
Philippe Gosset
Sandra M. Martín-Guerrero
Gábor M. Mórotz
Joseph Atherton
Jacqueline C. Mitchell
Andrea Markovinovic
Christopher C. J. Miller
Structural and functional studies of the VAPB-PTPIP51 ER-mitochondria tethering proteins in neurodegenerative diseases
Acta Neuropathologica Communications
Neurodegenerative diseases
Frontotemporal dementia
Amyotrophic lateral sclerosis
Alzheimer’s disease
Parkinson’s disease
title Structural and functional studies of the VAPB-PTPIP51 ER-mitochondria tethering proteins in neurodegenerative diseases
title_full Structural and functional studies of the VAPB-PTPIP51 ER-mitochondria tethering proteins in neurodegenerative diseases
title_fullStr Structural and functional studies of the VAPB-PTPIP51 ER-mitochondria tethering proteins in neurodegenerative diseases
title_full_unstemmed Structural and functional studies of the VAPB-PTPIP51 ER-mitochondria tethering proteins in neurodegenerative diseases
title_short Structural and functional studies of the VAPB-PTPIP51 ER-mitochondria tethering proteins in neurodegenerative diseases
title_sort structural and functional studies of the vapb ptpip51 er mitochondria tethering proteins in neurodegenerative diseases
topic Neurodegenerative diseases
Frontotemporal dementia
Amyotrophic lateral sclerosis
Alzheimer’s disease
Parkinson’s disease
url https://doi.org/10.1186/s40478-025-01964-7
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