Inhibition of insulin/IGF‐1 receptor signaling protects from mitochondria‐mediated kidney failure

Abstract Mitochondrial dysfunction and alterations in energy metabolism have been implicated in a variety of human diseases. Mitochondrial fusion is essential for maintenance of mitochondrial function and requires the prohibitin ring complex subunit prohibitin‐2 (PHB2) at the mitochondrial inner mem...

Full description

Saved in:
Bibliographic Details
Main Authors: Christina Ising, Sybille Koehler, Sebastian Brähler, Carsten Merkwirth, Martin Höhne, Olivier R Baris, Henning Hagmann, Martin Kann, Francesca Fabretti, Claudia Dafinger, Wilhelm Bloch, Bernhard Schermer, Andreas Linkermann, Jens C Brüning, Christine E Kurschat, Roman‐Ulrich Müller, Rudolf J Wiesner, Thomas Langer, Thomas Benzing, Paul Thomas Brinkkoetter
Format: Article
Language:English
Published: Springer Nature 2015-02-01
Series:EMBO Molecular Medicine
Subjects:
Online Access:https://doi.org/10.15252/emmm.201404916
Tags: Add Tag
No Tags, Be the first to tag this record!
_version_ 1849234875150761984
author Christina Ising
Sybille Koehler
Sebastian Brähler
Carsten Merkwirth
Martin Höhne
Olivier R Baris
Henning Hagmann
Martin Kann
Francesca Fabretti
Claudia Dafinger
Wilhelm Bloch
Bernhard Schermer
Andreas Linkermann
Jens C Brüning
Christine E Kurschat
Roman‐Ulrich Müller
Rudolf J Wiesner
Thomas Langer
Thomas Benzing
Paul Thomas Brinkkoetter
author_facet Christina Ising
Sybille Koehler
Sebastian Brähler
Carsten Merkwirth
Martin Höhne
Olivier R Baris
Henning Hagmann
Martin Kann
Francesca Fabretti
Claudia Dafinger
Wilhelm Bloch
Bernhard Schermer
Andreas Linkermann
Jens C Brüning
Christine E Kurschat
Roman‐Ulrich Müller
Rudolf J Wiesner
Thomas Langer
Thomas Benzing
Paul Thomas Brinkkoetter
author_sort Christina Ising
collection DOAJ
description Abstract Mitochondrial dysfunction and alterations in energy metabolism have been implicated in a variety of human diseases. Mitochondrial fusion is essential for maintenance of mitochondrial function and requires the prohibitin ring complex subunit prohibitin‐2 (PHB2) at the mitochondrial inner membrane. Here, we provide a link between PHB2 deficiency and hyperactive insulin/IGF‐1 signaling. Deletion of PHB2 in podocytes of mice, terminally differentiated cells at the kidney filtration barrier, caused progressive proteinuria, kidney failure, and death of the animals and resulted in hyperphosphorylation of S6 ribosomal protein (S6RP), a known mediator of the mTOR signaling pathway. Inhibition of the insulin/IGF‐1 signaling system through genetic deletion of the insulin receptor alone or in combination with the IGF‐1 receptor or treatment with rapamycin prevented hyperphosphorylation of S6RP without affecting the mitochondrial structural defect, alleviated renal disease, and delayed the onset of kidney failure in PHB2‐deficient animals. Evidently, perturbation of insulin/IGF‐1 receptor signaling contributes to tissue damage in mitochondrial disease, which may allow therapeutic intervention against a wide spectrum of diseases.
format Article
id doaj-art-aaed36cafd0a43ab8e7e6120f044f0e0
institution Kabale University
issn 1757-4676
1757-4684
language English
publishDate 2015-02-01
publisher Springer Nature
record_format Article
series EMBO Molecular Medicine
spelling doaj-art-aaed36cafd0a43ab8e7e6120f044f0e02025-08-20T04:03:00ZengSpringer NatureEMBO Molecular Medicine1757-46761757-46842015-02-017327528710.15252/emmm.201404916Inhibition of insulin/IGF‐1 receptor signaling protects from mitochondria‐mediated kidney failureChristina Ising0Sybille Koehler1Sebastian Brähler2Carsten Merkwirth3Martin Höhne4Olivier R Baris5Henning Hagmann6Martin Kann7Francesca Fabretti8Claudia Dafinger9Wilhelm Bloch10Bernhard Schermer11Andreas Linkermann12Jens C Brüning13Christine E Kurschat14Roman‐Ulrich Müller15Rudolf J Wiesner16Thomas Langer17Thomas Benzing18Paul Thomas Brinkkoetter19Department II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneInstitute for Genetics, University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneCenter for Physiology and Pathophysiology, Institute for Vegetative Physiology, University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneDepartment of Molecular and Cellular Sport Medicine, Institute of Cardiovascular Research and Sport Medicine, German Sport University CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneDivision of Nephrology and Hypertension, Christian‐Albrechts‐UniversityCologne Cluster of Excellence on Cellular Stress Responses in Ageing‐Associated Diseases (CECAD) and Systems Biology of Ageing Cologne (Sybacol), University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneCenter for Physiology and Pathophysiology, Institute for Vegetative Physiology, University of CologneInstitute for Genetics, University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneDepartment II of Internal Medicine and Center for Molecular Medicine Cologne (CMMC), University of CologneAbstract Mitochondrial dysfunction and alterations in energy metabolism have been implicated in a variety of human diseases. Mitochondrial fusion is essential for maintenance of mitochondrial function and requires the prohibitin ring complex subunit prohibitin‐2 (PHB2) at the mitochondrial inner membrane. Here, we provide a link between PHB2 deficiency and hyperactive insulin/IGF‐1 signaling. Deletion of PHB2 in podocytes of mice, terminally differentiated cells at the kidney filtration barrier, caused progressive proteinuria, kidney failure, and death of the animals and resulted in hyperphosphorylation of S6 ribosomal protein (S6RP), a known mediator of the mTOR signaling pathway. Inhibition of the insulin/IGF‐1 signaling system through genetic deletion of the insulin receptor alone or in combination with the IGF‐1 receptor or treatment with rapamycin prevented hyperphosphorylation of S6RP without affecting the mitochondrial structural defect, alleviated renal disease, and delayed the onset of kidney failure in PHB2‐deficient animals. Evidently, perturbation of insulin/IGF‐1 receptor signaling contributes to tissue damage in mitochondrial disease, which may allow therapeutic intervention against a wide spectrum of diseases.https://doi.org/10.15252/emmm.201404916insulinmitochondriamTORpodocyte
spellingShingle Christina Ising
Sybille Koehler
Sebastian Brähler
Carsten Merkwirth
Martin Höhne
Olivier R Baris
Henning Hagmann
Martin Kann
Francesca Fabretti
Claudia Dafinger
Wilhelm Bloch
Bernhard Schermer
Andreas Linkermann
Jens C Brüning
Christine E Kurschat
Roman‐Ulrich Müller
Rudolf J Wiesner
Thomas Langer
Thomas Benzing
Paul Thomas Brinkkoetter
Inhibition of insulin/IGF‐1 receptor signaling protects from mitochondria‐mediated kidney failure
EMBO Molecular Medicine
insulin
mitochondria
mTOR
podocyte
title Inhibition of insulin/IGF‐1 receptor signaling protects from mitochondria‐mediated kidney failure
title_full Inhibition of insulin/IGF‐1 receptor signaling protects from mitochondria‐mediated kidney failure
title_fullStr Inhibition of insulin/IGF‐1 receptor signaling protects from mitochondria‐mediated kidney failure
title_full_unstemmed Inhibition of insulin/IGF‐1 receptor signaling protects from mitochondria‐mediated kidney failure
title_short Inhibition of insulin/IGF‐1 receptor signaling protects from mitochondria‐mediated kidney failure
title_sort inhibition of insulin igf 1 receptor signaling protects from mitochondria mediated kidney failure
topic insulin
mitochondria
mTOR
podocyte
url https://doi.org/10.15252/emmm.201404916
work_keys_str_mv AT christinaising inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT sybillekoehler inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT sebastianbrahler inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT carstenmerkwirth inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT martinhohne inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT olivierrbaris inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT henninghagmann inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT martinkann inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT francescafabretti inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT claudiadafinger inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT wilhelmbloch inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT bernhardschermer inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT andreaslinkermann inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT jenscbruning inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT christineekurschat inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT romanulrichmuller inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT rudolfjwiesner inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT thomaslanger inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT thomasbenzing inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure
AT paulthomasbrinkkoetter inhibitionofinsulinigf1receptorsignalingprotectsfrommitochondriamediatedkidneyfailure