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Record W4416758536 · doi:10.1038/s41467-025-65308-8

Deleterious variants in the autophagy-related gene RB1CC1/FIP200 impair immunity to SARS-CoV-2

2025· article· en· W4416758536 on OpenAlexaff
Renée M. van der Sluis, Kennith Brian Castelino, Bao‐cun Zhang, Andreas Ronit, Thomas Zillinger, Marvin Werner, Sofie E. Jørgensen, Anne Louise Hansen, Alice Pedersen, Ryo Narita, Line S. Reinert, Bettina Bundgaard, Alessandro Aiuti, Saleh Al‐Muhsen, Fahd Al‐Mulla, Mark S. Anderson, Evangelos Andreakos, Andrés A. Arias, Lisa M. Arkin, Hagit Baris Feldman, Paul Bastard, Alexandre Belot, Catherine M. Biggs, Alexandre Bolze, Анастасія Бондаренко, A. Borghesi, Ahmed Aziz Bousfiha, Petter Brodin, Yenan T. Bryceson, Giorgio Casari, John Christodoulou, Aurélie Cobat, Roger Colobrán, Antônio Condino‐Neto, Stefan N. Constantinescu, Megan A. Cooper, Clifton L. Dalgard, Murkesh Desai, Beth A. Drolet, Munis Dündar, Xavier Duval, Sotiriјa Duvlis, Jamila El Baghdadi, Philippine Eloy, Sara Elva Espinosa‐Padilla, Jacques Fellay, Carlos Flores, José Luis Franco, Antoine Froidure, Guy Gorochov, Filomeen Haerynck, David Hagin, Rabih Halwani, Lennart Hammarström, James R. Heath, Elena W.Y. Hsieh, Eystein S. Husebye, Kohsuke Imai, Yuval Itan, Emmanuelle Jouanguy, Elżbieta Kaja, Timokratis Karamitros, Kai Kisand, Cheng‐Lung Ku, YL Lau, Yun Ling, C. Lucas, Matthieu Mahévas, Davood Mansouri, László Maródi, France Mentré, Isabelle Meyts, Joshua D. Milner, Kristina Mironska, Tomohiro Morio, Lisa F. P. Ng, L. D. Notarangelo, Antonio Novelli, Giuseppe Novelli, Cliona O’Farrelly, Satoshi Okada, Keisuke Okamoto, Tayfun Özçelık, Fırat Özçelik, Qiang Pan‐Hammarström, Rebeca Pérez de Diego, Jordi Pérez‐Tur, David S. Perlin, Jonny Peter, Anna M. Planas, Carolina Prando, Aurora Pujol, Anne Puel, Lluis Quintana-Murci, Sathishkumar Ramaswamy, Laurent Rénia, Igor Resnick, Carlos Rodríguez‐Gallego, Vanessa Sancho‐Shimizu, Anna Šedivá, Mikko Seppänen, Mohammad Shahrooei, Anna Shcherbina, Ondřej Slabý, Andrew L. Snow, Pere Soler‐Palacín, Vassili Soumelis, András N. Spaan, Helen C. Su, Ivan Tancevski, Stuart G. Tangye, Ahmad Abou Tayoun, Şehime Gülsün Temel, Christian W. Thorball, Pierre Tiberghien, Sophie Trouillet‐Assant, Stuart E. Turvey, K. M. Furkan Uddin, Mohammed Uddin, Diederik van de Beek, Fernanda Sales Luiz Vianna, Donald C. Vinh, Horst von Bernuth, Joost Wauters, Mayana Zatz, Qian Zhang, Shen‐Ying Zhang, Jacob Bodilsen, Kristoffer Skaalum Hansen, Merete Storgaard, Thomas Benfield, Marie Helleberg, Christian K. Holm, Jean‐Laurent Casanova, Fulvio Reggiori, Muriel Mari, Søren R. Paludan, Trine H. Mogensen

Bibliographic record

VenueNature Communications · 2025
Typearticle
Languageen
FieldMedicine
TopicAutophagy in Disease and Therapy
Canadian institutionsMcGill University Health CentreGenome CanadaUniversity of British ColumbiaBC Children's HospitalSt. Paul's Hospital
FundersH. Lundbeck A/SStavros Niarchos FoundationNovo NordiskFondation pour la Recherche MédicaleNational Institutes of HealthFisher Center for Alzheimer's Research FoundationSchweizerischer Nationalfonds zur Förderung der Wissenschaftlichen ForschungLundbeckfondenFondation du SouffleInstitut National de la Santé et de la Recherche MédicaleNovo Nordisk FondenInstitut des maladies génétiques ImagineNational Research FoundationMinistère de l'Enseignement supérieur, de la Recherche et de l'InnovationNational Institute of Allergy and Infectious DiseasesDanmarks GrundforskningsfondCHIST-ERANational Center for Advancing Translational SciencesHORIZON EUROPE Framework ProgrammeAgence Nationale de la RechercheGeorgia Clinical and Translational Science AllianceSt. Giles Foundation
KeywordsAutophagyGeneImmunityFunction (biology)InterferonViral replicationRNAHEK 293 cellsInnate immune system

Abstract

fetched live from OpenAlex

The clinical outcome of SARS-CoV-2 infection spans from asymptomatic viral elimination to lethal COVID-19 pneumonia, which is due to type I interferon (IFN) deficiency in at least 15–20% of cases. We report two unrelated male patients with critical COVID-19 who are heterozygous for rare deleterious variants in RB1CC1, encoding the autophagy-related FIP200 protein. Airway epithelial cells genetically deprived of FIP200 or cell lines expressing the RB1CC1/FIP200 patient variants exhibit elevated SARS-CoV-2 replication and impaired autophagic flux. The antiviral function of FIP200 is independent of canonical autophagy and type I IFN, but involves the selective autophagy receptor NDP52. We identify a non-canonical function of FIP200 in a novel lysosomal degradation pathway, in which SARS-CoV-2 virions are targeted to single-membrane compartments for degradation of viral RNA in LC3B-positive acidified vesicles. This pathway is impaired in FIP200-deficient cells and in cells expressing FIP200 patient haplotypes. Collectively, we describe a cell-autonomous anti-SARS-CoV-2 restriction pathway, dependent on FIP200 and NDP52, and independent of canonical autophagy and type I IFN, which can underlie critical COVID-19 pneumonia. The variability in clinical outcomes of SARS-CoV-2 infection is partly due to deficiencies in production or response to type I interferons (IFN). Here, the authors describe a FIP200-dependent lysosomal degradation pathway, independent of canonical autophagy and type I IFN, that restricts SARS-CoV-2 replication, offering insights into critical COVID-19 pneumonia mechanisms.

Fetched live from OpenAlex and de-inverted. Abstracts are not stored in this database: the inverted indexes are 8.6 GB of the frame’s 9.3 GB of text, and the host has 13 GB free.

How this classification was reachedexpand

Full frame machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.000
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: Bench or experimental
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.001
Threshold uncertainty score0.004

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0000.000
Insufficient payload (model declined to judge)0.0010.000

Machine scores (provisional)

The two teacher heads of the student model, read on this work. A score orders the frame for review; it never asserts a category, and the validation status ships verbatim with every row.

Baseline scores from an immature model (maturity gate not passed, 7 training rounds). Scores rank; they never assert a category.

Opus teacher head0.022
GPT teacher head0.360
Teacher spread0.339 · how far apart the two teachers sit on this one work
Validation statusscore_only:v0-immature-baseline · verbatim from the scoring run: score_only means the number may rank works, and no category label ships from it

Classification

machine, unvalidated

Machine predicted; a candidate call from one source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
Study designBench or experimental
Domainnot available
GenreEmpirical

How this classification was reached, model by model and score by score, is at the end of the page under "How this classification was reached".

Quick stats

Citations3
Published2025
Admission routes1
Has abstractyes

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