Center for Medical Ethics and Health Policy Staff Publications

Authors

Hyoungjun Ham
Huie Jing
Ian T Lamborn
Megan M Kober
Alexey Koval
Yamina A Berchiche
D Eric Anderson
Kirk M Druey
Judith N Mandl
Bertrand Isidor
Carlos R Ferreira
Alexandra F Freeman
Sundar Ganesan
Meliha Karsak
Peter J Mustillo
Juliana Teo
Zarazuela Zolkipli-Cunningham
Nicolas Chatron
François Lecoquierre
Andrew J Oler
Jana Pachlopnik Schmid
Douglas B Kuhns
Xuehua Xu
Fabian Hauck
Waleed Al-Herz
Matias Wagner
Paulien A Terhal
Mari Muurinen
Vincent Barlogis
Phillip Cruz
Jeffrey Danielson
Helen Stewart
Petra Loid
Sebastian Rading
Boris Keren
Rolph Pfundt
Kol A Zarember
Katharina Vill
Lorraine Potocki
Kenneth N Olivier
Gaetan Lesca
Laurence Faivre
Melanie Wong
Anne Puel
Janet Chou
Maud Tusseau
Niki M Moutsopoulos
Helen F Matthews
Cas Simons
Ryan J Taft
Ariane Soldatos
Etienne Masle-Farquhar
Stefania Pittaluga
Robert Brink
Danielle L Fink
Heidi H Kong
Juraj Kabat
Woo Sung Kim
Tatjana Bierhals
Kazuyuki Meguro
Amy P Hsu
Jingwen Gu
Jennifer Stoddard
Benito Banos-Pinero
Maria Slack
Giampaolo Trivellin
Benoît Mazel
Maarja Soomann
Samuel Li
Val J Watts
Constantine A Stratakis
Maria F Rodriguez-Quevedo
Ange-Line Bruel
Marita Lipsanen-Nyman
Paul Saultier
Rashmi Jain
Daphne Lehalle
Daniel Torres
Kathleen E Sullivan
Sébastien Barbarot
Axel Neu
Yannis Duffourd
Morgan Similuk
Kirsty McWalter
Pierre Blanc
Stéphane Bézieau
Tian Jin
Raif S Geha
Jean-Laurent Casanova
Outi M Makitie
Christian Kubisch
Patrick Edery
John Christodoulou
Ronald N Germain
Christopher C Goodnow
Thomas P Sakmar
Daniel D Billadeau
Sébastien Küry
Vladimir L Katanaev
Yu Zhang
Michael J Lenardo
Helen C Su

Language

English

Publication Date

9-20-2024

Journal

Science

DOI

10.1126/science.add8947

PMID

39298586

PMCID

PMC11811912

PubMedCentral® Posted Date

3-20-2025

PubMedCentral® Full Text Version

Author MSS

Abstract

Humans with monogenic inborn errors responsible for extreme disease phenotypes can reveal essential physiological pathways. We investigated germline mutations in GNAI2, which encodes Gαi2, a key component in heterotrimeric G-protein signal transduction usually thought to regulate adenylyl cyclase-mediated cAMP production. Patients with activating Gαi2 mutations had clinical presentations that included impaired immunity. Mutant Gαi2 impaired cell migration and augmented responses to T cell receptor (TCR) stimulation. We found that mutant Gαi2 influenced TCR signaling by sequestering the GTPase-activating protein RASA2, thereby promoting RAS activation and increasing downstream ERK/MAPK and PI3K-AKT S6 signaling to drive cellular growth and proliferation.

Keywords

Humans, Cell Movement, Cell Proliferation, Germ-Line Mutation, GTP-Binding Protein alpha Subunit, Gi2, Immunity, MAP Kinase Signaling System, Phosphatidylinositol 3-Kinases, Proto-Oncogene Proteins c-akt, ras GTPase-Activating Proteins, ras Proteins, Receptors, Antigen, T-Cell, Signal Transduction, T-Lymphocytes, Pedigree

Published Open-Access

yes

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