TANGO2 is an acyl-CoA binding protein

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  • dc.contributor.author Lujan, Agustin Leonardo
  • dc.contributor.author Foresti, Ombretta
  • dc.contributor.author Wojnacki, José
  • dc.contributor.author Bigliani, Gonzalo
  • dc.contributor.author Brouwers, Nathalie
  • dc.contributor.author Pena, Maria Jesus
  • dc.contributor.author Androulaki, Stefania
  • dc.contributor.author Hashidate-Yoshida, Tomomi
  • dc.contributor.author Kalyukina, Maria
  • dc.contributor.author Novoselov, Sergey S.
  • dc.contributor.author Shindou, Hideo
  • dc.contributor.author Malhotra, Vivek
  • dc.date.accessioned 2025-05-16T05:41:33Z
  • dc.date.available 2025-05-16T05:41:33Z
  • dc.date.issued 2025
  • dc.description.abstract Loss of TANGO2 in humans precipitates metabolic crises during periods of heightened energy demand, such as fasting, infections, or high fever. TANGO2 has been implicated in various functions, including lipid metabolism and heme transport, and its cellular localization remains uncertain. In our study, we demonstrate that TANGO2 localizes to the mitochondrial lumen via a structural region containing LIL residues. Mutations in these LIL residues cause TANGO2 to relocate to the periphery of lipid droplets. We further show that purified TANGO2 binds acyl-coenzyme A, and mutations in the highly conserved NRDE sequence of TANGO2 inhibit this binding. Collectively, our findings suggest that TANGO2 serves as an acyl-coenzyme A binding protein. These insights may provide new avenues for addressing the severe cardiomyopathies and rhabdomyolysis associated with defective TANGO2 in humans.
  • dc.format.mimetype application/pdf
  • dc.identifier.citation Lujan AL, Foresti O, Wojnacki J, Bigliani G, Brouwers N, Pena MJ, et al. TANGO2 is an acyl-CoA binding protein. J Cell Biol. 2025 May 5;224(5):e202410001. DOI: 10.1083/jcb.202410001
  • dc.identifier.doi http://dx.doi.org/10.1083/jcb.202410001
  • dc.identifier.issn 0021-9525
  • dc.identifier.uri http://hdl.handle.net/10230/70416
  • dc.language.iso eng
  • dc.publisher Rockefeller University Press
  • dc.relation.ispartof J Cell Biol. 2025 May 5;224(5):e202410001
  • dc.relation.projectID info:eu-repo/grantAgreement/EC/H2020/951146
  • dc.relation.projectID info:eu-repo/grantAgreement/ES/3PE/PID2022-143128NB-I00
  • dc.relation.projectID info:eu-repo/grantAgreement/EC/HE/101062382
  • dc.rights © 2025 Lujan et al. This work is licensed under a Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0).
  • dc.rights.accessRights info:eu-repo/semantics/openAccess
  • dc.rights.uri http://creativecommons.org/licenses/by/4.0
  • dc.subject.keyword Cell metabolism
  • dc.subject.keyword Disease
  • dc.title TANGO2 is an acyl-CoA binding protein
  • dc.type info:eu-repo/semantics/article
  • dc.type.version info:eu-repo/semantics/publishedVersion

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