Synergistic activation of the human phosphate exporter XPR1 by KIDINS220 and inositol pyrophosphate

Abstract Inorganic phosphate (Pi) is essential for life, and its intracellular levels must be tightly regulated to avoid toxicity. XPR1, the sole known phosphate exporter, is critical for maintaining this balance. Here we report cryo-EM structures of the human XPR1-KIDINS220 complex in substrate-fre...

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Main Authors: Peng Zuo, Weize Wang, Zonglin Dai, Jiye Zheng, Shang Yu, Guangxi Wang, Yue Yin, Ling Liang, Yuxin Yin
Format: Article
Language:English
Published: Nature Portfolio 2025-03-01
Series:Nature Communications
Online Access:https://doi.org/10.1038/s41467-025-58200-y
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author Peng Zuo
Weize Wang
Zonglin Dai
Jiye Zheng
Shang Yu
Guangxi Wang
Yue Yin
Ling Liang
Yuxin Yin
author_facet Peng Zuo
Weize Wang
Zonglin Dai
Jiye Zheng
Shang Yu
Guangxi Wang
Yue Yin
Ling Liang
Yuxin Yin
author_sort Peng Zuo
collection DOAJ
description Abstract Inorganic phosphate (Pi) is essential for life, and its intracellular levels must be tightly regulated to avoid toxicity. XPR1, the sole known phosphate exporter, is critical for maintaining this balance. Here we report cryo-EM structures of the human XPR1-KIDINS220 complex in substrate-free closed and substrate-bound outward-open states, as well as an XPR1 mutant in a substrate-bound inward-facing state. In the presence of inositol hexaphosphate (InsP6) and phosphate, the complex adopts an outward-open conformation, with InsP6 binding the SPX domain and juxtamembrane regions, indicating active phosphate export. Without phosphate or InsP6, the complex closes, with transmembrane helix 9 blocking the outward cavity and a C-terminal loop obstructing the intracellular cavity. XPR1 alone remains closed even with phosphate and InsP6. Functional mutagenesis shows that InsP6, whose levels vary with Pi availability, works with KIDINS220 to regulate XPR1 activity. These insights into phosphate regulation may aid in developing therapies for ovarian cancer.
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issn 2041-1723
language English
publishDate 2025-03-01
publisher Nature Portfolio
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series Nature Communications
spelling doaj-art-e43e732f452241ef80e37d65fd1e00b22025-08-20T03:40:48ZengNature PortfolioNature Communications2041-17232025-03-0116111310.1038/s41467-025-58200-ySynergistic activation of the human phosphate exporter XPR1 by KIDINS220 and inositol pyrophosphatePeng Zuo0Weize Wang1Zonglin Dai2Jiye Zheng3Shang Yu4Guangxi Wang5Yue Yin6Ling Liang7Yuxin Yin8Institute of Systems Biomedicine, Department of Pathology, Beijing Key Laboratory of Tumor Systems Biology, School of Basic Medical Sciences, Peking University Health Science CenterInstitute of Systems Biomedicine, Department of Pathology, Beijing Key Laboratory of Tumor Systems Biology, School of Basic Medical Sciences, Peking University Health Science CenterInstitute of Systems Biomedicine, Department of Pathology, Beijing Key Laboratory of Tumor Systems Biology, School of Basic Medical Sciences, Peking University Health Science CenterDepartment of Biophysics, School of Basic Medical Sciences, Peking University Health Science CenterDepartment of Biophysics, School of Basic Medical Sciences, Peking University Health Science CenterInstitute of Systems Biomedicine, Department of Pathology, Beijing Key Laboratory of Tumor Systems Biology, School of Basic Medical Sciences, Peking University Health Science CenterDepartment of Pharmacology, School of Basic Medical Sciences, Peking University Health Science CenterInstitute of Systems Biomedicine, Department of Pathology, Beijing Key Laboratory of Tumor Systems Biology, School of Basic Medical Sciences, Peking University Health Science CenterInstitute of Systems Biomedicine, Department of Pathology, Beijing Key Laboratory of Tumor Systems Biology, School of Basic Medical Sciences, Peking University Health Science CenterAbstract Inorganic phosphate (Pi) is essential for life, and its intracellular levels must be tightly regulated to avoid toxicity. XPR1, the sole known phosphate exporter, is critical for maintaining this balance. Here we report cryo-EM structures of the human XPR1-KIDINS220 complex in substrate-free closed and substrate-bound outward-open states, as well as an XPR1 mutant in a substrate-bound inward-facing state. In the presence of inositol hexaphosphate (InsP6) and phosphate, the complex adopts an outward-open conformation, with InsP6 binding the SPX domain and juxtamembrane regions, indicating active phosphate export. Without phosphate or InsP6, the complex closes, with transmembrane helix 9 blocking the outward cavity and a C-terminal loop obstructing the intracellular cavity. XPR1 alone remains closed even with phosphate and InsP6. Functional mutagenesis shows that InsP6, whose levels vary with Pi availability, works with KIDINS220 to regulate XPR1 activity. These insights into phosphate regulation may aid in developing therapies for ovarian cancer.https://doi.org/10.1038/s41467-025-58200-y
spellingShingle Peng Zuo
Weize Wang
Zonglin Dai
Jiye Zheng
Shang Yu
Guangxi Wang
Yue Yin
Ling Liang
Yuxin Yin
Synergistic activation of the human phosphate exporter XPR1 by KIDINS220 and inositol pyrophosphate
Nature Communications
title Synergistic activation of the human phosphate exporter XPR1 by KIDINS220 and inositol pyrophosphate
title_full Synergistic activation of the human phosphate exporter XPR1 by KIDINS220 and inositol pyrophosphate
title_fullStr Synergistic activation of the human phosphate exporter XPR1 by KIDINS220 and inositol pyrophosphate
title_full_unstemmed Synergistic activation of the human phosphate exporter XPR1 by KIDINS220 and inositol pyrophosphate
title_short Synergistic activation of the human phosphate exporter XPR1 by KIDINS220 and inositol pyrophosphate
title_sort synergistic activation of the human phosphate exporter xpr1 by kidins220 and inositol pyrophosphate
url https://doi.org/10.1038/s41467-025-58200-y
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