The mTOR-Dop1a-Agpat2 axis regulates nuclear phospholipid homeostasis.
Overview
and 26 other authors
Mukhtar Ullah14,15, Mathieu Quinodoz14,15,16, Carlo Rivolta14,15,16, Abdul Ghafoor Khan17, Muhammad Nadeem Khan17, Muhammad Ansar18,19, Erica H Gerkes20, Tuula Rinne21, Alexander PA Stegmann22, Margje Sinnema22, Malak Ali Alghamdi23, Essa Alharby24, Reham M Balahmar24, Naif AM Almontashiri24,25, Sarah Baer26,27, Amélie Piton28,26, Carla Díes Curià29, Sandra Mercier30,31, Benjamin Cogné30,31, Patrick Yap32,33, Shin-ya Morita34, Akiyoshi Kakita35,36, Mitsuhiro Kato37,38, Yoshihiro Maruo2, Naomichi Matsumoto3,4,39,40, Masaki Mori1,2,640 affiliations
- Department of Pediatric Physiology, Innovation & Research Support Center, Graduate School, International University of Health and Welfare, Tokyo, Japan
- Department of Pediatrics, Shiga University of Medical Science, Otsu, Japan
- Department of Human Genetics, Yokohama City University Graduate School of Medicine, Yokohama, Japan
- Department of Clinical Genetics, Yokohama City University Hospital, Yokohama, Japan
- Department of Neurogenetics, Molecular Neuroscience Research Center, Shiga University of Medical Science, Otsu, Japan
- Advanced Pediatric Medicine, Tohoku University School of Medicine, Miyagi, Japan
- Department of Reproductive Medicine, Center for Regenerative Medicine, National Center for Child Health and Development (NCCHD), Tokyo, Japan
- Research Center for Animal Life Science (RCALS), Shiga University of Medical Science, Otsu, Japan
- Department of Biochemistry, Yokohama City University Graduate School of Medicine, Yokohama, Japan
- Department of Child Neurology, National Center Hospital, National Center of Neurology and Psychiatry, Tokyo, Japan
- Department of Pediatrics, Nara Medical University, Kashihara, Japan
- Omics Research Center, National Cerebral and Cardiovascular Center, Osaka, Japan
- Department of Advanced Medical Technologies, National Cerebral and Cardiovascular Center, Osaka, Japan
- Institute of Molecular and Clinical Ophthalmology Basel, Basel, Switzerland
- Department of Ophthalmology, University of Basel, Basel, Switzerland
- Department of Genetics and Genome Biology, University of Leicester, Leicester, UK
- Bannu Medical College, MTI Bannu, Bannu, Pakistan
- Department of Ophthalmology, University of Lausanne, Jules-Gonin Eye Hospital, Fondation Asile des aveugles, Lausanne, Switzerland
- Advanced Molecular Genetics and Genomics Disease Research and Treatment Centre, Dow University of Health Sciences, Karachi, Pakistan
- University of Groningen, University Medical Center Groningen, Department of Genetics, Nijmegen, the Netherlands
- Department of Human Genetics, Donders Institute for Brain, Cognition and Behavior, Radboud University Medical Center, Nijmegen, the Netherlands
- Department of Clinical Genetics, Maastricht University Medical Center, Maastricht, the Netherlands
- Medical Genetics Division, Pediatric Department, College of Medicine, King Saud University, Riyadh, Saudi Arabia
- Center for Genetics and Inherited Diseases, Taibah University, Almadinah Almunwarah, Saudi Arabia
- Faculty of Applied Medical Sciences, Taibah University, Almadinah Almunwarah, Saudi Arabia
- Institut de Génétique et de Biologie Moléculaire et Cellulaire, Illkirch, France
- Department of Pediatric Neurology, Strasbourg University Hospital, Strasbourg, France
- Laboratoire de Diagnostic Génétique, Institut de Génétique Médicale d'Alsace (IGMA), Hôspitaux Universitaire de Strasbourg, Strasbourg, France
- IMPaCT-Genómica - Enfermedades Raras, Servei de Genètica, Hospital del Mar, Universitat Pompeu Fabra (UPF), Barcelona, Spain
- Service de génétique médicale, CHU de Nantes, Nantes, France
- Institut du thorax, INSERM, CNRS, Nantes Université, Nantes, France
- Genetic Health Service New Zealand (Northern Hub), Auckland City Hospital, University of Auckland, Auckland, New Zealand
- Faculty of Medicine and Health Sciences, University of Auckland, Auckland, New Zealand
- Department of Pharmacotherapeutics, Shiga University of Medical Science, Otsu, Japan
- Department of Pathology, Brain Research Institute, Niigata University, Niigata, Japan
- Center for Human Brain Resource Initiative (ChBRI), Niigata University, Niigata, Japan
- Department of Pediatrics, Showa Medical University, Tokyo, Japan
- Epilepsy Medical Center, Showa Medical University Hospital, Tokyo, Japan
- Department of Rare Disease Genomics, Yokohama City University Hospital, Yokohama, Japan
- Medical Genome Center, National Center of Neurology and Psychiatry, Kodaira, Japan
Abstract
The molecular mechanisms regulating the phospholipid (PL) metabolism in the nucleus remain to be elucidated. Here, we describe the role of Dop1a in controlling PL abundance in nuclear membranes (NMs) under the control of mTOR signaling. A shortage of lysophosphatidic acid (LPA) triggers the rapid localization of Dop1a to the nuclear pore complexes (NPCs), where Dop1a suppresses PL synthesis by binding to AGPAT2 (1-acylglycerol-3-phosph
Reproduced under the paper's license (CC BY), from the paper cited above.
Code
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The paper's code and data availability statement is in the Data section.
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Data
No dataset and no data link were found in the paper.
Data and code availability
The scRNA-seq data generated in this study have been deposited in the DNA Data Bank of Japan (DDBJ) database under the BioProject accession number PRJDB40458 and experiment accession number E-GEAD-1221. Additional datasets are available from the lead contact upon reasonable request.
Reproduced under the paper's license (CC BY), from the paper cited above.
Versions
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Version 1, 29 September 2026: the first record
Recorded: type, language, journal, volume, issue, pages, dates, 46 authors, 1 keyword, 8 funders, 58 references, 2 RRIDs.
Cite
This paper
Ariyama, H., Tsukamura, A., Miyatake, S., Okado, S., Itabashi, I., Ogura, A., Suzuki, A., Kurakawa, H., Sakaguchi, Y., Nakatake, Y., Sanada, R., Terakado, I., Koshimizu, E., Mizuguchi, T., Hamada, K., Ogata, K., Nakagawa, E., Sakakibara, T., Shirai, M., . . . Mori, M. (2026). The mTOR-Dop1a-Agpat2 axis regulates nuclear phospholipid homeostasis. iScience, 29(6), 115860. https://
BibTeX
@article{ariyama2026mtor
author = {Ariyama, Hirotaka and Tsukamura, Atsushi and Miyatake, Satoko and Okado, Satoko and Itabashi, Itsuki and Ogura, Ami and Suzuki, Atsunobu and Kurakawa, Hyuga and Sakaguchi, Yuki and Nakatake, Yuhki and Sanada, Ryunosuke and Terakado, Ichiro and Koshimizu, Eriko and Mizuguchi, Takeshi and Hamada, Keisuke and Ogata, Kazuhiro and Nakagawa, Eiji and Sakakibara, Takafumi and Shirai, Manabu and Fujihara, Yoshitaka and Ullah, Mukhtar and Quinodoz, Mathieu and Rivolta, Carlo and Khan, Abdul Ghafoor and Khan, Muhammad Nadeem and Ansar, Muhammad and Gerkes, Erica H and Rinne, Tuula and Stegmann, Alexander PA and Sinnema, Margje and Alghamdi, Malak Ali and Alharby, Essa and Balahmar, Reham M and Almontashiri, Naif AM and Baer, Sarah and Piton, Amélie and Curià, Carla Díes and Mercier, Sandra and Cogné, Benjamin and Yap, Patrick and Morita, Shin-ya and Kakita, Akiyoshi and Kato, Mitsuhiro and Maruo, Yoshihiro and Matsumoto, Naomichi and Mori, Masaki},
title = {{The mTOR-Dop1a-Agpat2 axis regulates nuclear phospholipid homeostasis}},
journal = {iScience},
year = {2026},
month = apr,
volume = {29},
number = {6},
pages = {115860},
publisher = {Elsevier},
issn = {2589-0042},
doi = {10.1016/
url = {https://
pmid = {42164854},
pmcid = {PMC13186030}
}
RIS
TY - JOUR
AU - Ariyama, Hirotaka
AU - Tsukamura, Atsushi
AU - Miyatake, Satoko
AU - Okado, Satoko
AU - Itabashi, Itsuki
AU - Ogura, Ami
AU - Suzuki, Atsunobu
AU - Kurakawa, Hyuga
AU - Sakaguchi, Yuki
AU - Nakatake, Yuhki
AU - Sanada, Ryunosuke
AU - Terakado, Ichiro
AU - Koshimizu, Eriko
AU - Mizuguchi, Takeshi
AU - Hamada, Keisuke
AU - Ogata, Kazuhiro
AU - Nakagawa, Eiji
AU - Sakakibara, Takafumi
AU - Shirai, Manabu
AU - Fujihara, Yoshitaka
AU - Ullah, Mukhtar
AU - Quinodoz, Mathieu
AU - Rivolta, Carlo
AU - Khan, Abdul Ghafoor
AU - Khan, Muhammad Nadeem
AU - Ansar, Muhammad
AU - Gerkes, Erica H
AU - Rinne, Tuula
AU - Stegmann, Alexander PA
AU - Sinnema, Margje
AU - Alghamdi, Malak Ali
AU - Alharby, Essa
AU - Balahmar, Reham M
AU - Almontashiri, Naif AM
AU - Baer, Sarah
AU - Piton, Amélie
AU - Curià, Carla Díes
AU - Mercier, Sandra
AU - Cogné, Benjamin
AU - Yap, Patrick
AU - Morita, Shin-ya
AU - Kakita, Akiyoshi
AU - Kato, Mitsuhiro
AU - Maruo, Yoshihiro
AU - Matsumoto, Naomichi
AU - Mori, Masaki
TI - The mTOR-Dop1a-Agpat2 axis regulates nuclear phospholipid homeostasis
T2 - iScience
J2 - iScience
PY - 2026
DA - 2026/
VL - 29
IS - 6
SP - 115860
SN - 2589-0042
PB - Elsevier
DO - 10.1016/
UR - https://
LA - en
ER -
CSL-JSON
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