[go: up one dir, main page]
More Web Proxy on the site http://driver.im/
Skip to main content

Thank you for visiting nature.com. You are using a browser version with limited support for CSS. To obtain the best experience, we recommend you use a more up to date browser (or turn off compatibility mode in Internet Explorer). In the meantime, to ensure continued support, we are displaying the site without styles and JavaScript.

  • Article
  • Published:

NUFIP1 integrates amino acid sensing and DNA damage response to maintain the intestinal homeostasis

Abstract

Nutrient availability strongly affects intestinal homeostasis. Here, we report that low-protein (LP) diets decrease amino acids levels, impair the DNA damage response (DDR), cause DNA damage and exacerbate inflammation in intestinal tissues of male mice with inflammatory bowel disease (IBD). Intriguingly, loss of nuclear fragile X mental retardation-interacting protein 1 (NUFIP1) contributes to the amino acid deficiency-induced impairment of the DDR in vivo and in vitro and induces necroptosis-related spontaneous enteritis. Mechanistically, phosphorylated NUFIP1 binds to replication protein A2 (RPA32) to recruit the ataxia telangiectasia and Rad3-related (ATR)–ATR-interacting protein (ATRIP) complex, triggering the DDR. Consistently, both reintroducing NUFIP1 but not its non-phospho-mutant and inhibition of necroptosis prevent bowel inflammation in male Nufip1 conditional knockout mice. Intestinal inflammation and DNA damage in male mice with IBD can be mitigated by NUFIP1 overexpression. Moreover, NUFIP1 protein levels in the intestine of patients with IBD were found to be significantly decreased. Conclusively, our study uncovers that LP diets contribute to intestinal inflammation by hijacking NUFIP1–DDR signalling and thereby activating necroptosis.

This is a preview of subscription content, access via your institution

Access options

Buy this article

Prices may be subject to local taxes which are calculated during checkout

Fig. 1: LP diets incite intestinal DNA damage and inflammation.
Fig. 2: NUFIP1 is essential to the amino acid-regulated DDR.
Fig. 3: Loss of NUFIP1 triggers spontaneous enteritis.
Fig. 4: Phosphorylation of NUFIP1 determines its function in the DDR.
Fig. 5: NUFIP1 recruits the ATR–ATRIP complex to DNA damage sites.
Fig. 6: Loss of NUFIP1 or amino acid deficiency enhances necroptosis.
Fig. 7: Upregulation of NUFIP1 alleviates intestinal inflammation.

Similar content being viewed by others

Data availability

Phosphorylation sites were found in the protein PTM database (https://www.phosphosite.org). The mass spectrometry proteomics data have been deposited to the ProteomeXchange Consortium via the PRIDE partner repository with the dataset identifier PXD053976. The dataset GSE111889 was downloaded from the GEO database45 (https://www.ncbi.nlm.nih.gov/geo/). Source data are provided with this paper.

Code availability

No custom codes were used in this study.

References

  1. Di Francesco, A., Di Germanio, C., Bernier, M. & de Cabo, R. A time to fast. Science 362, 770–775 (2018).

    Article  PubMed  PubMed Central  Google Scholar 

  2. Rubio-Patino, C. et al. Low-protein diet induces IRE1α-dependent anticancer immunosurveillance. Cell Metab. 27, 828–842 (2018).

    Article  PubMed  CAS  Google Scholar 

  3. Kelly, B. & Pearce, E. L. Amino assets: how amino acids support immunity. Cell Metab. 32, 154–175 (2020).

    Article  PubMed  CAS  Google Scholar 

  4. Buck, M. D., Sowell, R. T., Kaech, S. M. & Pearce, E. L. Metabolic instruction of immunity. Cell 169, 570–586 (2017).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  5. Sies, H., Berndt, C. & Jones, D. P. Oxidative stress. Annu. Rev. Biochem. 86, 715–748 (2017).

    Article  PubMed  CAS  Google Scholar 

  6. Nilsson, R. et al. Metabolic enzyme expression highlights a key role for MTHFD2 and the mitochondrial folate pathway in cancer. Nat. Commun. 5, 3128 (2014).

    Article  PubMed  Google Scholar 

  7. Ericksen, R. E. et al. Loss of BCAA catabolism during carcinogenesis enhances mTORC1 activity and promotes tumor development and progression. Cell Metab. 29, 1151–1165 (2019).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  8. Saxton, R. A. & Sabatini, D. M. mTOR signaling in growth, metabolism, and disease. Cell 169, 361–371 (2017).

    Article  PubMed  CAS  Google Scholar 

  9. Kwon, N. H. et al. Dual role of methionyl-tRNA synthetase in the regulation of translation and tumor suppressor activity of aminoacyl-tRNA synthetase-interacting multifunctional protein-3. Proc. Natl Acad. Sci. USA 108, 19635–19640 (2011).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  10. Jackson, S. P. & Bartek, J. The DNA-damage response in human biology and disease. Nature 461, 1071–1078 (2009).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  11. Lopez-Otin, C., Blasco, M. A., Partridge, L., Serrano, M. & Kroemer, G. The hallmarks of aging. Cell 153, 1194–1217 (2013).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  12. Osborn, A. J., Elledge, S. J. & Zou, L. Checking on the fork: the DNA-replication stress-response pathway. Trends Cell Biol. 12, 509–516 (2002).

    Article  PubMed  CAS  Google Scholar 

  13. Shimizu, I., Yoshida, Y., Suda, M. & Minamino, T. DNA damage response and metabolic disease. Cell Metab. 20, 967–977 (2014).

    Article  PubMed  CAS  Google Scholar 

  14. Thomson, G. J. et al. Metabolism-induced oxidative stress and DNA damage selectively trigger genome instability in polyploid fungal cells. EMBO J. 38, e101597 (2019).

    Article  PubMed  PubMed Central  Google Scholar 

  15. Hu, C. M. et al. High glucose triggers nucleotide imbalance through O-GlcNAcylation of key enzymes and induces KRAS mutation in pancreatic cells. Cell Metab. 29, 1334–1349 (2019).

    Article  PubMed  CAS  Google Scholar 

  16. Fang, E. F. et al. NAD+ replenishment improves lifespan and healthspan in ataxia telangiectasia models via mitophagy and DNA repair. Cell Metab. 24, 566–581 (2016).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  17. Xie, Y. et al. Gut epithelial TSC1/mTOR controls RIPK3-dependent necroptosis in intestinal inflammation and cancer. J. Clin. Invest. 130, 2111–2128 (2020).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  18. Rass, U., Ahel, I. & West, S. C. Defective DNA repair and neurodegenerative disease. Cell 130, 991–1004 (2007).

    Article  PubMed  CAS  Google Scholar 

  19. Zhao, Y., Simon, M., Seluanov, A. & Gorbunova, V. DNA damage and repair in age-related inflammation. Nat. Rev. Immunol. 23, 75–89 (2022).

  20. Blackford, A. N. & Jackson, S. P. ATM, ATR, and DNA-PK: the trinity at the heart of the DNA damage response. Mol. Cell 66, 801–817 (2017).

    Article  PubMed  CAS  Google Scholar 

  21. Ciccia, A. & Elledge, S. J. The DNA damage response: making it safe to play with knives. Mol. Cell 40, 179–204 (2010).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  22. Zou, L. & Elledge, S. J. Sensing DNA damage through ATRIP recognition of RPA–ssDNA complexes. Science 300, 1542–1548 (2003).

    Article  PubMed  CAS  Google Scholar 

  23. Kim, W. et al. ZFP161 regulates replication fork stability and maintenance of genomic stability by recruiting the ATR/ATRIP complex. Nat. Commun. 10, 5304 (2019).

    Article  PubMed  PubMed Central  Google Scholar 

  24. Cortez, D., Guntuku, S., Qin, J. & Elledge, S. J. ATR and ATRIP: partners in checkpoint signaling. Science 294, 1713–1716 (2001).

    Article  PubMed  CAS  Google Scholar 

  25. Hanada, K. et al. The structure-specific endonuclease Mus81 contributes to replication restart by generating double-strand DNA breaks. Nat. Struct. Mol. Biol. 14, 1096–1104 (2007).

    Article  PubMed  CAS  Google Scholar 

  26. Harper, J. W. & Elledge, S. J. The DNA damage response: ten years after. Mol. Cell 28, 739–745 (2007).

    Article  PubMed  CAS  Google Scholar 

  27. Bardoni, B. et al. NUFIP1 (nuclear FMRP interacting protein 1) is a nucleocytoplasmic shuttling protein associated with active synaptoneurosomes. Exp. Cell Res. 289, 95–107 (2003).

    Article  PubMed  CAS  Google Scholar 

  28. Wyant, G. A. et al. NUFIP1 is a ribosome receptor for starvation-induced ribophagy. Science 360, 751–758 (2018).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  29. Bizarro, J. et al. Proteomic and 3D structure analyses highlight the C/D box snoRNP assembly mechanism and its control. J. Cell Biol. 207, 463–480 (2014).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  30. Gowen, L. C., Avrutskaya, A. V., Latour, A. M., Koller, B. H. & Leadon, S. A. BRCA1 required for transcription-coupled repair of oxidative DNA damage. Science 281, 1009–1012 (1998).

    Article  PubMed  CAS  Google Scholar 

  31. Hatchi, E. et al. BRCA1 recruitment to transcriptional pause sites is required for R-loop-driven DNA damage repair. Mol. Cell 57, 636–647 (2015).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  32. Cabart, P., Chew, H. K. & Murphy, S. BRCA1 cooperates with NUFIP and P-TEFb to activate transcription by RNA polymerase II. Oncogene 23, 5316–5329 (2004).

    Article  PubMed  CAS  Google Scholar 

  33. Alpatov, R. et al. A chromatin-dependent role of the fragile X mental retardation protein FMRP in the DNA damage response. Cell 157, 869–881 (2014).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  34. Zhang, X. et al. High-protein diets increase cardiovascular risk by activating macrophage mTOR to suppress mitophagy. Nat. Metab. 2, 110–125 (2020).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  35. Chatzidoukaki, O. et al. R-loops trigger the release of cytoplasmic ssDNAs leading to chronic inflammation upon DNA damage. Sci. Adv. 7, eabj5769 (2021).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  36. Ge, M. K. et al. The tRNA–GCN2–FBXO22-axis-mediated mTOR ubiquitination senses amino acid insufficiency. Cell Metab. 35, 2216–2230 (2023).

    Article  PubMed  CAS  Google Scholar 

  37. Sugiyama, Y. & Uezato, Y. Analysis of protein kinases by Phos-tag SDS–PAGE. J. Proteomics 255, 104485 (2022).

    Article  PubMed  CAS  Google Scholar 

  38. Cano-Crespo, S. et al. CD98hc (SLC3A2) sustains amino acid and nucleotide availability for cell cycle progression. Sci. Rep. 9, 14065 (2019).

    Article  PubMed  PubMed Central  Google Scholar 

  39. Byun, T. S., Pacek, M., Yee, M. C., Walter, J. C. & Cimprich, K. A. Functional uncoupling of MCM helicase and DNA polymerase activities activates the ATR-dependent checkpoint. Genes Dev. 19, 1040–1052 (2005).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  40. Ciccia, A. et al. The SIOD disorder protein SMARCAL1 is an RPA-interacting protein involved in replication fork restart. Genes Dev. 23, 2415–2425 (2009).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  41. Mer, G. et al. Structural basis for the recognition of DNA repair proteins UNG2, XPA, and RAD52 by replication factor RPA. Cell 103, 449–456 (2000).

    Article  PubMed  CAS  Google Scholar 

  42. Patankar, J. V. & Becker, C. Cell death in the gut epithelium and implications for chronic inflammation. Nat. Rev. Gastroenterol. Hepatol. 17, 543–556 (2020).

    Article  PubMed  Google Scholar 

  43. Sun, L. et al. Mixed lineage kinase domain-like protein mediates necrosis signaling downstream of RIP3 kinase. Cell 148, 213–227 (2012).

    Article  PubMed  CAS  Google Scholar 

  44. Wang, R. et al. Gut stem cell necroptosis by genome instability triggers bowel inflammation. Nature 580, 386–390 (2020).

    Article  PubMed  CAS  Google Scholar 

  45. Lloyd-Price, J. et al. Multi-omics of the gut microbial ecosystem in inflammatory bowel diseases. Nature 569, 655–662 (2019).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  46. Ko, G. J., Rhee, C. M., Kalantar-Zadeh, K. & Joshi, S. The effects of high-protein diets on kidney health and longevity. J. Am. Soc. Nephrol. 31, 1667–1679 (2020).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  47. Wolfson, R. L. & Sabatini, D. M. The dawn of the age of amino acid sensors for the mTORC1 pathway. Cell Metab. 26, 301–309 (2017).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  48. Xie, X. et al. The mTOR–S6K pathway links growth signalling to DNA damage response by targeting RNF168. Nat. Cell Biol. 20, 320–331 (2018).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  49. Jiao, H. et al. Z-nucleic-acid sensing triggers ZBP1-dependent necroptosis and inflammation. Nature 580, 391–395 (2020).

    Article  PubMed  PubMed Central  CAS  Google Scholar 

  50. Chassaing, B., Aitken, J. D., Malleshappa, M. & Vijay-Kumar, M. Dextran sulfate sodium (DSS)-induced colitis in mice. Curr. Protoc. Immunol. 104, 15.25.11–15.25.14 (2014).

    Article  Google Scholar 

  51. Grieger, J. C., Choi, V. W. & Samulski, R. J. Production and characterization of adeno-associated viral vectors. Nat. Protoc. 1, 1412–1428 (2006).

    Article  PubMed  CAS  Google Scholar 

  52. Sato, T. et al. Single Lgr5 stem cells build crypt–villus structures in vitro without a mesenchymal niche. Nature 459, 262–265 (2009).

    Article  PubMed  CAS  Google Scholar 

  53. Li, M. L. et al. S-adenosyl-l-methionine supplementation alleviates damaged intestinal epithelium and inflammatory infiltration caused by Mat2a deficiency. Development 150, dev201135 (2023).

    Article  PubMed  CAS  Google Scholar 

  54. Tummala, K. S. et al. Inhibition of de novo NAD+ synthesis by oncogenic URI causes liver tumorigenesis through DNA damage. Cancer Cell 26, 826–839 (2014).

    Article  PubMed  CAS  Google Scholar 

  55. Henricksen, L. A., Umbricht, C. B. & Wold, M. S. Recombinant replication protein A: expression, complex formation, and functional characterization. J. Biol. Chem. 269, 11121–11132 (1994).

    Article  PubMed  CAS  Google Scholar 

  56. Qian, L. et al. Enhanced BCAT1 activity and BCAA metabolism promotes RhoC activity in cancer progression. Nat. Metab. 5, 1159–1173 (2023).

    Article  PubMed  CAS  Google Scholar 

Download references

Acknowledgements

We thank members of the Lei laboratory for discussion throughout this study and the Biomedical Core Facility of Fudan University for technical support. We thank S. Bing (Shanghai Institute of Immunology, Department of Immunology and Microbiology, Shanghai Jiao Tong University School of Medicine, Shanghai 200025, China) for offering the ΔDEPDC5 stable HeLa cell line. This work was supported by the National Key R&D Program of China (2020YFA0803402 and 2019YFA0801703 to Q.-Y.L.), the Natural Science Foundation of China (82121004, 82330092 and 81790250 to Q.-Y.L.; 82103116 to H.M.; 82472873 to M.Y.), the Innovation Program of Shanghai Municipal Education Commission (2023ZKZD11), the Shanghai Municipal Science and Technology Major Project and the New Cornerstone Science Foundation (to Q.-Y.L.).

Author information

Authors and Affiliations

Authors

Contributions

H.M., J.T., M.Y. and Q.-Y.L. designed experiments, performed data analyses and wrote the manuscript. H.M. and J.T. performed the most experiments. S.-Y.C., C.-P.Y., Y.-T.Q., C.W., Y.L., L.Z. and J.Y. helped with the methodology. Q.-Y.L. and M.Y. supervised the study.

Corresponding authors

Correspondence to Miao Yin or Qun-Ying Lei.

Ethics declarations

Competing interests

The authors declare no competing interests.

Peer review

Peer review information

Nature Metabolism thanks David S. Yu and the other, anonymous, reviewer(s) for their contribution to the peer review of this work. Primary Handling Editor: Yanina-Yasmin Pesch, in collaboration with the Nature Metabolism team.

Additional information

Publisher’s note Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Extended data

Extended Data Fig. 1 Low-protein (LP) diets aggravate intestinal DNA damage and inflammation.

a, Representative IHC images of colon tissues from mice fed with AIN-93G, 6% protein, or 60% protein (high-protein, HP) diets and water containing 2% DSS. b, Quantification of IHC of γH2A.X in crypts of colon tissues from mice fed with AIN-93G or 60% protein diets and water containing 2% DSS. n = 73 (AIN-93G) and 69 (60% protein) crypts. 3 independent experiments. c, Quantification of IHC of inflammatory cells markers in colon tissues from mice in (b). Sample size (n) see SourceData_Extended_Fig1. d, Representative IHC images of p-ATR and γH2A.X in colon tissues from mice fed with AIN-93G or LP diets. DDR was induced by intraperitoneal injection of 5-FU. n = 3 mice. e, Western blot of p-CHK1 and γH2A.X in HeLa cells in response to HU treatment and deprivation of individual amino acid. n = 3 independent experiments. f, Relative quantification of p-CHK1 in HeLa cells in response to HU treatment and deprivation of individual amino acid. Normal culture medium as control. n = 3 independent experiments. g, Relative quantification of γH2A.X level in HeLa cells in response to HU treatment and deprivation of individual amino acid. Normal culture medium as control. n = 3 independent experiments. h, Western blot of p-ATR, p-CHK1 and γH2A.X in HeLa cells. 2 mM HU was added to control or GCN2 knockout HeLa cells with or without amino acids starvation. n = 3 independent experiments. i, j, Knockout of GCN1 (i) (n = 3 independent experiments.) or DEPDC5 (j) (n = 3 independent experiments) does not rescue the levels of DDR markers downregulated by amino acids deprivation in HeLa cells. Western blot analyses of levels of p-ATR, p-CHK1 and γH2A.X in cells in response to HU treatment with or without amino acids starvation. Scale bar, 50 µm. Error bars represented mean ± s.d. Statistical comparisons were made using Mann–Whitney U-test (b, c) or one-way ANOVA test (f, g, h, j). NS, not significant.

Source data

Extended Data Fig. 2 NUFIP1 is essential for amino acid-regulated DDR.

a, Gene Ontology (GO) analysis of biological processes of chromatin-bound proteins in HeLa cells. The selected proteins were enriched more than 1.5 times in HU treatment group than in NC group. The enriched terms are ranked by -log10 (p values). n = 1. b, c, Western blot of p-ATR, p-CHK1 and γH2A.X in NUFIP1 knockdown (b) and knockout (c) HeLa cells. Cells were treated with 2 mM HU. n = 3 independent experiments. d, e, Representative IHC images of p-ATR (d) and γH2A.X (e) in colon tissues from mice fed with AIN-93G or LP diets. EV or NUFIP1 was overexpressed in colon tissues of mice fed with LP diets using AAV delivery system. DDR was induced by 5-FU treatment. Scale bars, 50 µm. n = 4 mice. Error bars represented mean ± s.d. p values were determined by one-way ANOVA test.

Source data

Extended Data Fig. 3 The loss of NUFIP1 triggers spontaneous enteritis.

a, b, Mouse model (a) and schematic representation (b). One dose (400 mg/kg) of tamoxifen was used. c, NUFIP1 is deleted in intestinal epithelium Nufip1(Δ/Δ) iIEC mice. Western blot of NUFIP1 protein level in ileum and colon tissues from Nufip1fl/fl, Nufip1(+/Δ) iIEC and Nufip1(Δ/Δ) iIEC mice at 7 dpi of tamoxifen treatment. Representative results were from two independent mice. d, Nufip1fl/fl, Nufip1(+/Δ) iIEC and Nufip1(Δ/Δ) iIEC mice weight change after tamoxifen treatment. Data were represented as mean±s.d. p values were derived from two-way ANOVA test. n = 6 mice. e, Representative IHC images of Ki-67 and γH2A.X in ileum tissues from Nufip1fl/fl and Nufip1(Δ/Δ) iIEC mice at 7dpi of tamoxifen treatment. n = 6 mice. f, Representative IHC images of Ki-67 and γH2A.X in colon tissues from Nufip1fl/fl and Nufip1(Δ/Δ) iIEC mice at 7dpi of tamoxifen treatment. n = 6 mice. g, Representative IHC images of inflammatory cells markers in ileum tissues from Nufip1fl/fl and Nufip1(Δ/Δ) iIEC mice at 7 dpi of tamoxifen treatment. n = 6 mice. h, Representative IHC images of inflammatory cells markers in colon tissues of Nufip1fl/fl and Nufip1(Δ/Δ) iIEC mice at 7 dpi of tamoxifen treatment. n = 6 mice. i, Western blot analysis of NUFIP1 in ileum and colon tissues from Nufip1fl/fl, Nufip1(+/Δ) iIEC and Nufip1(Δ/Δ) iIEC mice at 28 dpi of tamoxifen treatment. Representative results were from two independent mice. j, Representative H&E staining images of ileum and colon sections from Nufip1fl/fl and Nufip1(Δ/Δ) iIEC mice at 28 dpi of tamoxifen treatment. n = 3 mice. Scale bar, 50 µm.

Source data

Extended Data Fig. 4 Phosphorylation of NUFIP1 determines its function on DDR signaling.

a, Phos-tag blot analysis of NUFIP1. n = 3 independent experiments. b, Phos-tag blot analysis of NUFIP1WT, NUFIP1S292A, NUFIP1S403A and NUFIP1S292/403A. n = 3 independent experiments. c, Anti-p-NUFIP1 (S292) antibody specifically recognizes site-specific (S292) phospho-peptide of NUFIP1. n = 3 independent experiments. d, NUFIP1S292A mutant decreases phosphorylation level of p-NUFIP1. n = 3 independent experiments. e, Phos-tag blot analysis of NUFIP1 in HeLa cells treated with kinase inhibitors. No. 1 VE822. No. 2 AZ20. No. 3 RGB-286638. No. 4 AZD-5438. No. 5 THZ531. No. 6 Ralimetinib dimesylate. No. 7 AZ304. No. 8 PF-04691502. No.9 CLK-IN-T3. n = 3 independent experiments. f, Phos-tag blot analysis of NUFIP1WT, NUFIP1S292A, or NUFIP1S403A in HeLa cells treated with RGB-286638 or AZD-5438. n = 3 independent experiments. g, Detection of p-S292 level using anti-pS292 antibody after overexpression of kinases. FLAG-NUFIP1 was obtained from HEK293T cells by immunoprecipitation. n = 3 independent experiments. h, Quantification of p-NUFIP1 after overexpression kinases. i, Detection of NUFIP1 p-S292 level with p-S292 antibody after overexpression of CDK9WT or CDK9D167N. n = 3 independent experiments. j, Western blot of p-NUFIP1 based on in vitro kinase assay. n = 3 independent experiments. k, Western blot of p-NUFIP1, p-ATR, p-CHK1 and γH2A.X in HeLa cells. Control and CDK9 knockdown cells were treated with HU. n = 3 independent experiments. l, CDK9 protein level in chromatin fraction. n = 3 independent experiments. m, Phos-tag blot analysis of NUFIP1WT and NUFIP1S283A. n = 3 independent experiments. n, Colon tissue was obtained from Nufip1fl/fl and AAV-infected Nufip1(Δ/Δ) iIEC mice at 7dpi of tamoxifen treatment. Representative results were from two independent mice. o, Representative IHC images of Ki-67 and γH2A.X in colon tissue from mice in (n). n = 6 mice. p, Representative IHC images of inflammatory cells markers in colon tissues from mice in (n). n = 6 mice. Scale bar, 50 µm. The data were represented as mean±s.d. p values in (e, h, i, k, l) were determined by one-way ANOVA test. p values in (j) were determined by two-tailed unpaired t-test.

Source data

Extended Data Fig. 5 LC3B-interacting regions (LIRs) of NUFIP1 is irrelevant to its function on DDR regulation.

a, Western blot of p-ATR, p-CHK1 and γH2A.X in HEK293T cells. 2 mM HU was added to cells with or without amino acids starvation. n = 3 independent experiments. b, Schematic representation of AAV-infected Nufip1(Δ/Δ) iIEC mice. Mice was fed with 6% protein diets and DDR was induced by 5-FU treatment. c, Proteins were isolated from colon tissue of mice in (b) and examined with indicated antibodies. Representative results were from three independent mice. d, The representative IHC images of p-ATR and γH2A.X in colon tissues from mice in (b). n = 3 mice. e, Quantification of IHC of p-ATR and γH2A.X in colonic crypts from mice in (b). n = 61 crypts. 3 independent experiments. Scale bar, 50 µm. The data were represented as mean±s.d. Statistical comparisons were made using one-way ANOVA test (a) or Kolmogorov-Smirnov test (e).

Source data

Extended Data Fig. 6 NUFIP1 facilitates the recruitment of ATRIP/ATR complex to chromatin upon DNA damage.

a, Chromatin fraction was obtained from normal or NUFIP1 knockdown HeLa cells treated with HU. Proteins were detected using indicated antibodies. n = 3 independent experiments. b, Chromatin fraction was obtained from NUFIP1-silenced HeLa cells with NUFIP1WT or NUFIP1S292A putback. Proteins were detected with indicated antibodies. n = 3 independent experiments. c,d, Representative immunofluorescent staining images of ATR (c) and ATRIP (d) in normal or NUFIP1 knockdown HeLa cells treated with HU. The nucleus was circled by the dashed line. RPA32 as the negative control. Blue, DAPI. Green, RPA32. Red, ATR/ATRIP. Scale bar, 10 µm. n = 3 independent experiments. e, Proteins were obtained from HU-treated HeLa cells overexpressing FLAG-NUFIP1 by immunoprecipitation and examined using indicated antibodies. FLAG-HNRNPU as the negative control. n = 3 independent experiments.

Source data

Extended Data Fig. 7 Loss of NUFIP1 or amino acids deficiency enhances necroptosis in intestine.

a, Representative images and statistical analysis of immunofluorescent staining of Cleaved Caspase-3 (CC3) in ileum and colon tissues from Nufip1fl/fl and Nufip1(Δ/Δ) iIEC mice at 7-dpi. Blue, DAPI. Red, CC3. Sample size (n) see SourceData_Extended_Fig7. b, PI tracing of intestinal organoids isolated from Villin-creERT2 Nufip1fl/fl mice. Organoids were treated with 4-OHT in presence or absence of zVAD-fmk, as indicated. The right panel is the statistical analysis of relative PI area per organoid (n = 3 independent experiments. Representative results are from 9 organoids). c, PI tracing was performed with intestinal organoid from Villin-creETR2 Nufip1fl/fl mice. Organoids were treated with 4-OHT in presence or absence of GSK′872, as indicated. The right panel is the statistical analysis of relative PI area per organoid (n = 3 independent experiments. Representative results are from 9 organoids). d, Western blot of RIPK3. Proteins from colon tissues of AAV infected Nufip1(Δ/Δ) iIEC mice were examined using the indicated antibodies. Representative results were from three independent mice. e, Representative IHC images of p-RIPK3 and p-MLKL in colon tissues from AAV-infected Nufip1(Δ/Δ) iIEC mice. Area within the black box was the enlarged view of dashed line area. n = 3 mice. f, Representative IHC images of inflammatory cells markers in colon tissues from AAV-infected Nufip1(Δ/Δ) iIEC mice. Area within black box was the enlarged view of dashed line area. n = 3 mice. g, Representative images of TUNEL assays in colon tissues of mice fed with AIN-93G or 6% protein diets and water containing 2% DSS. Area within the white box was the enlarged view of dashed line area. n = 3 mice. h, Representative IHC images of p-MLKL and p-RIPK3 in colon tissues from mice in (g). Area within the black box was the enlarged view of dashed line area. The red arrowheads denoted dead cells. n = 3 mice. Scale bar, 50 µm. Data were represented as mean ± s.d. Statistical comparisons were made using Mann–Whitney U-test (a) or one-way ANOVA test (b, c) or two-tailed unpaired t test (d).

Source data

Extended Data Fig. 8 Enhancing NUFIP1 expression alleviates intestinal inflammation in IBD mouse model.

a, Representative H&E and IHC images of colon tissues from mice infected with AAV and fed with water containing 1.5% DSS. Area within the black box was the enlarged view of dashed line area. Scale bar, 50 µm. n = 4 mice.

Supplementary information

Reporting Summary

Supplementary Table 1

Chromatin-associated protein detected by LC–MS.

Supplementary Table 2

Clinical sample information.

Supplementary Table 3

Antibodies.

Source data

Source Data Fig. 1

Statistical source data.

Source Data Fig. 1

Unprocessed western blots.

Source Data Fig. 2

Statistical source data.

Source Data Fig. 2

Unprocessed western blots.

Source Data Fig. 3

Statistical source data.

Source Data Fig. 4

Statistical source data.

Source Data Fig. 4

Unprocessed western blots.

Source Data Fig. 5

Statistical source data.

Source Data Fig. 5

Unprocessed western blots.

Source Data Fig. 6

Statistical source data.

Source Data Fig. 6

Unprocessed western blots.

Source Data Fig. 7

Statistical source data.

Source Data Fig. 7

Unprocessed western blots.

Source Data Extended Data Fig. 1

Statistical source data.

Source Data Extended Data Fig. 1

Unprocessed western blots.

Source Data Extended Data Fig. 2

Statistical source data.

Source Data Extended Data Fig. 2

Unprocessed western blots.

Source Data Extended Data Fig. 3

Statistical source data.

Source Data Extended Data Fig. 3

Unprocessed western blots.

Source Data Extended Data Fig. 4

Statistical source data.

Source Data Extended Data Fig. 4

Unprocessed western blots.

Source Data Extended Data Fig. 5

Statistical source data.

Source Data Extended Data Fig. 5

Unprocessed western blots.

Source Data Extended Data Fig. 6

Unprocessed western blots.

Source Data Extended Data Fig. 7

Statistical source data.

Source Data Extended Data Fig. 7

Unprocessed western blots.

Rights and permissions

Springer Nature or its licensor (e.g. a society or other partner) holds exclusive rights to this article under a publishing agreement with the author(s) or other rightsholder(s); author self-archiving of the accepted manuscript version of this article is solely governed by the terms of such publishing agreement and applicable law.

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Ming, H., Tan, J., Cao, SY. et al. NUFIP1 integrates amino acid sensing and DNA damage response to maintain the intestinal homeostasis. Nat Metab (2025). https://doi.org/10.1038/s42255-024-01179-5

Download citation

  • Received:

  • Accepted:

  • Published:

  • DOI: https://doi.org/10.1038/s42255-024-01179-5

Search

Quick links

Nature Briefing

Sign up for the Nature Briefing newsletter — what matters in science, free to your inbox daily.

Get the most important science stories of the day, free in your inbox. Sign up for Nature Briefing