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Paper · 20160352

Wolfram syndrome 1 gene negatively regulates ER stress signaling in rodent and human cells.

Fonseca SG and 10 others. J Clin Invest, 2010. Cited by 348 papers, by iCite.

11 sourcespapers-2026-11-12-0.1.0

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ConductScience Library. Wolfram syndrome 1 gene negatively regulates ER stress signaling in rodent and human cells. Papers, record 20160352. Release papers-2026-11-12-0.1.0, 12 Nov 2026. https://conductscience.org/library/papers/20160352

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Paper

PubMed's record of the paper, with iCite's citation figures. No abstract is shown.

Title
Wolfram syndrome 1 gene negatively regulates ER stress signaling in rodent and human cells.
PubMed
Journal
J Clin Invest
PubMed
Year
2010
PubMed
Publication types
Journal Article, Research Support, N.I.H., Extramural, Research Support, Non-U.S. Gov't
PubMed
Citations, by iCite
348
iCite
Relative citation ratio, by iCite
7.93
iCite
Revised in PubMed
2022-03-17
PubMed

Authors11

The authors in the order PubMed's record lists them, with the ORCID iDs it states.

OrderAuthorForenameKindORCID iD
1Fonseca SGSonya G
2Ishigaki SShinsuke
3Oslowski CMChristine M
4Lu SSimin
5Lipson KLKathryn L
6Ghosh RRajarshi
7Hayashi EEmiko
8Ishihara HHisamitsu
9Oka YYoshitomo
10Permutt MAM Alan

1 to 10 of 11Source PubMed

See all 11 authors

Topics21

NLM's MeSH indexing of the paper, its major topics first.

MeSH termMajor
Calmodulin-Binding ProteinsMajor topic
Insulin-Secreting CellsMajor topic
Membrane ProteinsMajor topic
Signal TransductionMajor topic
Unfolded Protein ResponseMajor topic
Wolfram SyndromeMajor topic
Activating Transcription Factor 6
Animals
COS Cells
Cell Line, Tumor
Chlorocebus aethiops
Endoplasmic Reticulum
Gene Expression Regulation
Humans
Mice
Proteasome Endopeptidase Complex
Rats
Trans-Activators
Ubiquitin
Ubiquitin-Protein Ligases
Ubiquitination

Source PubMed

Grants3

Each grant PubMed's record writes, matched to the NIH project NIH's grant and paper links name for this paper, and each of those links the record does not write.

Grant, as the record writes itAgencyCountryNIH projectStated bySource
P30 DK032520NIDDK NIH HHSUnited StatesP30DK032520PubMed and NIHNIH RePORTER
R01 DK067493NIDDK NIH HHSUnited StatesR01DK067493PubMed and NIHNIH RePORTER
5 P30 DK32520NIDDK NIH HHSUnited StatesP30DK032520PubMed and NIHNIH RePORTER

Trials0

The trials whose ClinicalTrials.gov record lists the paper, and the trial numbers the paper's PubMed record names.

None in this release.

Comments and corrections0

PubMed's comments and corrections, NLM's own indexing: retractions first, then expressions of concern, errata and updates, and comments last.

None in this release.

References45

The papers the reference list in PubMed's record names, those in our copy of PubMed, in the list's order.

OrderPMIDTitleJournalYearIn PapersSource
117,603,484Common variants in WFS1 confer risk of type 2 diabetes.Nat Genet2007https://conductscience.org/library/papers/17603484PubMed
216,215,705Mice conditionally lacking the Wolfram gene in pancreatic islet beta cells exhibit diabetes as a result of enhanced endoplasmic reticulum stress and apoptosis.Diabetologia2005https://conductscience.org/library/papers/16215705PubMed
312,586,069A time-dependent phase shift in the mammalian unfolded protein response.Dev Cell2003https://conductscience.org/library/papers/12586069PubMed
49,252,404Cytosolic degradation of T-cell receptor alpha chains by the proteasome.J Biol Chem1997https://conductscience.org/library/papers/9252404PubMed
515,056,606Disruption of the WFS1 gene in mice causes progressive beta-cell loss and impaired stimulus-secretion coupling in insulin secretion.Hum Mol Genet2004https://conductscience.org/library/papers/15056606PubMed
619,657,394A versatile viral system for expression and depletion of proteins in mammalian cells.PLoS One2009https://conductscience.org/library/papers/19657394PubMed
717,158,450
Selective inhibition of eukaryotic translation initiation factor 2 alpha dephosphorylation potentiates fatty acid-induced endoplasmic reticulum stress and causes pancreatic beta-cell dysfunction and apoptosis.
J Biol Chem2007https://conductscience.org/library/papers/17158450PubMed
810,856,300Activation of ATF6 and an ATF6 DNA binding site by the endoplasmic reticulum stress response.J Biol Chem2000https://conductscience.org/library/papers/10856300PubMed
99,817,917Diabetes insipidus, diabetes mellitus, optic atrophy and deafness (DIDMOAD) caused by mutations in a novel gene (wolframin) coding for a predicted transmembrane protein.Hum Mol Genet1998https://conductscience.org/library/papers/9817917PubMed
1015,211,570Endoplasmic reticulum stress triggers an acute proteasome-dependent degradation of ATF6.J Cell Biochem2004https://conductscience.org/library/papers/15211570PubMed

1 to 10 of 45

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Cited by266

The papers in our copy of PubMed whose reference list names this one, newest first. iCite, which reads more sources, counts 348 citations.

PMIDTitleJournalYearIn PapersSource
42,864,579The ubiquitin-proteasome system and proteostasis in the cochlea and sensorineural hearing disorders.Mol Biomed2026https://conductscience.org/library/papers/42864579PubMed
42,856,230Hyperexcitable but seizure-free: hippocampal network dysfunction in a Wolfram syndrome model.bioRxiv2026https://conductscience.org/library/papers/42856230PubMed
42,829,918WFS1 Deficiency Impairs PIAS4-Associated SUMOylation and Increases Ubiquitin-Mediated Spermatogenesis-Related Protein Degradation Leading to Testicular Male Infertility.Adv Sci (Weinh)2026https://conductscience.org/library/papers/42829918PubMed
42,773,111The anion channel GPR89 is a tumor-specific dependency in breast cancer.Nat Commun2026https://conductscience.org/library/papers/42773111PubMed
42,752,965Hepatic PDIA5 improves hyperglycaemia and insulin resistance through stabilisation of ATF6.Diabetologia2026https://conductscience.org/library/papers/42752965PubMed
42,651,706Deletion of wfs1 Impairs Oligodendrocyte Precursor Cells Dorsal Distribution and Myelination Through the wfs1-hmgcs1 Axis in Zebrafish.Biology (Basel)2026https://conductscience.org/library/papers/42651706PubMed
42,597,412Evaluating the use of GLP-1 receptor agonists in Wolfram syndrome patients.Front Endocrinol (Lausanne)2026https://conductscience.org/library/papers/42597412PubMed
42,524,523Genotype-based severity scoring system in Wolfram Syndrome: correlation with onset of cardinal symptoms and WFS1 gene variant types.Front Genet2026https://conductscience.org/library/papers/42524523PubMed
42,443,863Specialization of the upper and lower olfactory rosettes reveals sex- and origin-dependent plasticity in gene expression in Senegalese sole (Solea senegalensis).BMC Biol2026https://conductscience.org/library/papers/42443863PubMed
42,428,113Consensus Recommendations for the Clinical Management of Wolfram syndrome Using a Delphi Method.medRxiv2026https://conductscience.org/library/papers/42428113PubMed

1 to 10 of 266

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Genes4

The genes the Gene search release ties to the paper: Swiss-Prot's references first, then PubTator's text mining.

GeneNCBI Gene IDLinked byIn GenesSource
ATF622,926PubTator's text mininghttps://conductscience.org/library/genes/22926NCBI Gene
PSMA55,686PubTator's text mininghttps://conductscience.org/library/genes/5686NCBI Gene
SYVN184,447PubTator's text mininghttps://conductscience.org/library/genes/84447NCBI Gene
WFS17,466PubTator's text mininghttps://conductscience.org/library/genes/7466NCBI Gene

Drugs0

The drugs whose trials, in the Pipelines release, list the paper.

None in this release.

Data0

The data bank entries PubMed's record names. Trial numbers are under Trials.

None in this release.

Data dates

The date of each source this release read.

PubMed
2026-11-06
PubMed
iCite
2026-11-05
iCite
PMC's ID file
2026-11-05
PMC's ID file
NIH RePORTER's grant and paper links
2026-11-01
NIH RePORTER's grant and paper links
Trials
2026-11-12
Gene search
2026-11-11
Pipelines
2026-09-18

Sources 11

  • ChEMBL, through Open TargetsCC BY-SA 3.0
  • ClinicalTrials.govNo license stated
  • HGNCCC0
  • iCiteCC BY 4.0
  • NCBI GenePublic domain (US government)
  • NIH RePORTER's grant and paper linksPublic domain
  • Open TargetsCC0 1.0
  • PMC's ID filePublic domain (US government)
  • PubMedNLM's terms
  • PubTator 3Public domain
  • UniProtCC BY 4.0
Each source's terms
  • ChEMBL, through Open TargetsCC BY-SA 3.0, for the parts from ChEMBL. Read on 8 Nov 2026 at https://platform-docs.opentargets.org/licence
  • ClinicalTrials.govnone stated
  • HGNCCC0: https://www.genenames.org/about/license/
  • iCiteCC BY 4.0
  • NCBI GeneNone named for the file. NCBI says information created by or for the US government on its site is in the public domain, and asks that NLM be acknowledged: https://www.ncbi.nlm.nih.gov/home/about/policies/
  • NIH RePORTER's grant and paper linksPublic domain (NIH RePORTER)
  • Open TargetsCC0 1.0. Open Targets asks that users cite its latest paper: Buniello et al. 2025, Open Targets Platform: facilitating therapeutic hypotheses building in drug discovery, Nucleic Acids Research 53(D1):D1467, PMID 39657122. Read on 8 Nov 2026 at https://platform-docs.opentargets.org/licence
  • PMC's ID fileNone named for the file. NCBI says information created by or for the US government on its site is in the public domain, and asks that NLM be acknowledged: https://www.ncbi.nlm.nih.gov/home/about/policies/
  • PubMedNLM's terms
  • PubTator 3Public domain
  • UniProtCC BY 4.0. UniProt's licence file: "All databases and documents in the UniProt FTP directory may be copied and redistributed freely, without advance permission, provided that this copyright statement is reproduced with each copy." UniProt asks that users cite its latest paper: The UniProt Consortium, UniProt: the Universal Protein Knowledgebase in 2025, Nucleic Acids Research 53:D609-D617, https://doi.org/10.1093/nar/gkae1010. Read on 9 Nov 2026 at https://www.uniprot.org/help/license

ClinicalTrials.gov's data carry an international copyright outside the United States: https://clinicaltrials.gov/about-site/terms-conditions.

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