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Record W4405444871 · doi:10.1084/jem.20241413

A common form of dominant human IFNAR1 deficiency impairs IFN-α and -ω but not IFN-β-dependent immunity

2024· article· en· W4405444871 on OpenAlexaff
Fahd Al Qureshah, Jérémie Le Pen, Nicole A. de Weerd, Marcela Moncada‐Vélez, Marie Materna, Daniel Chieh-Ding Lin, Fernanda Sales Luiz Vianna, Lucy Bizien, Lazaro Lorenzo, Marc Lecuit, Jean‐David Pommier, Sevgi Keleş, Tayfun Özçelık, Sigifredo Pedraza‐Sánchez, Nicolas de Prost, Loubna El Zein, Hassan Hammoud, Lisa F. P. Ng, Rabih Halwani, Narjes Saheb Sharif‐Askari, YL Lau, Anthony Raymond Tam, Neha Singh, Sagar Bhattad, Yackov Berkun, Wasun Chantratita, Raúl Aguilar-López, Mohammad Shahrooei, Laurent Abel, Alessandro Aiuti, Saleh Al‐Muhsen, Ana Bertha Alcántara-Garduño, Evangelos Andreakos, Andrés A. Arias, Hagit Baris Feldman, Paul Bastard, Alexandre Bolze, A. Borghesi, Ahmed Aziz Bousfiha, Petter Brodin, John Christodoulou, Aurélie Cobat, Roger Colobrán, Antonio Condino-Neto, Sotiriјa Duvlis, Xavier Duval, Munis Dündar, Soha Fakhreddine, Jacques Fellay, Carlos Flores, José Luis Barrera Franco, Guy Gorochov, Peter K. Gregersen, David Hagin, María Teresa Herrera, Ivan Fan-Ngai Hung, Emmanuelle Jouanguy, Daniel Leung, Tom Le-voyer, Davood Mansouri, Jesús Mercado-García, Isabelle Meyts, Trine H. Mogensen, Antonio Novelli, Giuseppe Novelli, Satoshi Okada, Fırat Özçelik, Rebeca Pérez de Diego, Jordi Pérez‐Tur, Graziano Pesole, Anne Puel, Laurent Rénia, Igor Resnick, Carlos Rodríguez‐Gallego, Manal Sbeity, Sahar Sedighzadeh, Pere Soler‐Palacín, András N. Spaan, Stuart G. Tangye, Ahmad Abou Tayoun, Şehime Gülsün Temel, Christian Thorball, Ibrahim Torktaz, Sophie Trouillet‐Assant, Stuart E. Turvey, K. M. Furkan Uddin, Donald C. Vinh, Oscar Zabaleta-Martínez, Qian Zhang, Shen‐Ying Zhang, Jean‐Laurent Casanova, Chanreaksmey Eng, Kimrong Bun, MengHeng Oum, Patrice Piola, Arnaud Tarantola, Mey Channa, Veasna Duong, Philippe Buchy, Chris Gorman, Yoann Crabol, Philippe Dussart, M. Bunleat, M. Panha, Malcolm Sim, Em Bunnakea, Denis Laurent, Sothy Heng, Ky Santy, Anousone Douangnouvong, Danoy Chommanam, Khansoudaphone Phakhounthong, Manivanh Vongsouvath, Malee Seephone, Bountoy Sibounheunang, Sayaphet Rattanavong, Viengmon Davong, Malavanh Vongsouvath, Mayfong Mayxay, Audrey Dubot‐Pérès, Paul N. Newton, Sommanikhone Phangmanixay, Khounthavy Phongsavath, Đặng Đức Anh, Do Quyen, Tran Thi Mai Hung, Nguyễn Thị Thu Thủy, Luong Minh Tan, Anh Tuấn Thanh Phạm, Nguyễn Trần Hiển, Do Thu Huong, Lê Thanh Hải, Nguyễn Văn Lâm, Phạm Nhật An, Phan Hữu Phúc, Phung Bich Thuy, Trần Thị Thu Hương, Chaw Su Hlaing, Aye Mya Min Aye, Cho Thair, Kyaw Linn, May July, Win Thein, Latt Latt Kyaw, Htay Htay Tin, Ommar Swe Tin, Khin Yi Oo, Magali Herrant, Magali Lago, Maud Seguy, Marc Jouan, Lukas Hafner, Philippe Pérot, Marc Éloit, Olivier Lortholary, Julien Capelle, Bruno Rosset, Véronique Chevalier, Jérôme Honnorat, Anne Laurie Pinto, Xavier de Lamballerie, Kevin Bleakley, Bernadette Murgue, Catherine Ferrant, Christian Devaux, Hervé Tissot‐Dupont, Jean‐Paul Moatti, Pascal Bonnet, Didier Fontenille, Jean-François Delfraissy, Patrice Debré, Vivien Béziat, Peng Zhang, Charles M. Rice, Paul J. Hertzog

Bibliographic record

VenueThe Journal of Experimental Medicine · 2024
Typearticle
Languageen
FieldImmunology and Microbiology
Topicinterferon and immune responses
Canadian institutionsASTERSickKids FoundationHospital for Sick Children
FundersNIH Clinical CenterNational Institute of Allergy and Infectious DiseasesFisher Center for Alzheimer's Research FoundationAgence Nationale de la RechercheNational Center for Research ResourcesKing Abdulaziz City for Science and TechnologyStavros Niarchos FoundationKing Abdullah University of Science and TechnologyNational Center for Advancing Translational SciencesKing Saud UniversitySCOR Corporate Foundation for ScienceInstitut des maladies génétiques ImagineKarolinska InstitutetInstitut National de la Santé et de la Recherche MédicaleUniversity of Texas Southwestern Medical CenterFondation du SouffleWellcome TrustMeyer FoundationRockefeller UniversityNational Institutes of HealthAmerican Lung AssociationSt. Giles FoundationConselho Nacional de Desenvolvimento Científico e TecnológicoInstitut de Recherche pour le DéveloppementHoward Hughes Medical Institute
KeywordsBiologyAlleleDominance (genetics)HaploinsufficiencyGeneticsInterferonImmunityImmunologyPhenotypeHeterozygote advantageGeneImmune system

Abstract

fetched live from OpenAlex

Autosomal recessive deficiency of the IFNAR1 or IFNAR2 chain of the human type I IFN receptor abolishes cellular responses to IFN-α, -β, and -ω, underlies severe viral diseases, and is globally very rare, except for IFNAR1 and IFNAR2 deficiency in Western Polynesia and the Arctic, respectively. We report 11 human IFNAR1 alleles, the products of which impair but do not abolish responses to IFN-α and -ω without affecting responses to IFN-β. Ten of these alleles are rare in all populations studied, but the remaining allele (P335del) is common in Southern China (minor allele frequency ≈2%). Cells heterozygous for these variants display a dominant phenotype in vitro with impaired responses to IFN-α and -ω, but not -β, and viral susceptibility. Negative dominance, rather than haploinsufficiency, accounts for this dominance. Patients heterozygous for these variants are prone to viral diseases, attesting to both the dominance of these variants clinically and the importance of IFN-α and -ω for protective immunity against some viruses.

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 distilled prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. Learned from the 10,348 direct Codex labels and 10,348 direct Gemma labels. Candidate is the union of thresholded teacher heads; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels or direct frontier model labels.

metaresearch head score (Codex)0.001
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation 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 score0.078
Threshold uncertainty score0.610

Codex and Gemma teacher scores by category

CategoryCodexGemma
Metaresearch0.0010.000
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.000
Bibliometrics0.0000.000
Science and technology studies0.0000.001
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 head0.020
GPT teacher head0.317
Teacher spread0.297 · 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 teacher head, 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

Citations13
Published2024
Admission routes1
Has abstractyes

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