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Record W3211341335 · doi:10.1038/s41398-021-01680-5

A missense variant in SHARPIN mediates Alzheimer’s disease-specific brain damages

2021· article· en· W3211341335 on OpenAlexfundno aff
Jun Young Park, Dongsoo Lee, Jang Jae Lee, Jungsoo Gim, Tamil Iniyan Gunasekaran, Kyu Yeong Choi, Sarang Kang, Ah Ra, Jinyeon Jo, Juhong Park, Kyungtaek Park, Donghe Li, Sanghun Lee, Hoowon Kim, Immanuel Dhanasingh, Suparna Ghosh, Seula Keum, Jee Hye Choi, Gyun Jee Song, Lee Sael, Sangmyung Rhee, Simon Lovestone, Eunae Kim, Seung Hwan Moon, Byeong C. Kim, SangYun Kim, Andrew J. Saykin, Kwangsik Nho, Sung Haeng Lee, Lindsay A. Farrer, Gyungah Jun, Sungho Won, Kun Ho Lee

Bibliographic record

VenueTranslational Psychiatry · 2021
Typearticle
Languageen
FieldMedicine
TopicAlzheimer's disease research and treatments
Canadian institutionsnot available
FundersNational Institute on AgingNational Institute of Biomedical Imaging and BioengineeringCanadian Institutes of Health ResearchGenentechNational Institutes of HealthU.S. National Library of MedicineIXICOMinistry of Science and ICT, South KoreaH. Lundbeck A/SServierEisaiMeso Scale DiagnosticsNational Research Foundation of KoreaNational IT Industry Promotion AgencyNational Research FoundationKorea Health Industry Development InstituteNorthern California Institute for Research and EducationPfizerBiogenBioClinicaF. Hoffmann-La RocheUniversity of Southern CaliforniaEuropean CommissionBristol-Myers SquibbKorea Brain Research InstituteEli Lilly and CompanyU.S. Department of DefenseAlzheimer's Disease Neuroimaging InitiativeNovartis Pharmaceuticals CorporationAlzheimer's AssociationFoundation for the National Institutes of Health
KeywordsMissense mutationHippocampal formationEntorhinal cortexNeuroscienceAlzheimer's Disease Neuroimaging InitiativePathogenesisAlzheimer's diseasePsychologyDiseaseBiologyMedicineGeneticsInternal medicineMutationGene

Abstract

fetched live from OpenAlex

Abstract Established genetic risk factors for Alzheimer’s disease (AD) account for only a portion of AD heritability. The aim of this study was to identify novel associations between genetic variants and AD-specific brain atrophy. We conducted genome-wide association studies for brain magnetic resonance imaging measures of hippocampal volume and entorhinal cortical thickness in 2643 Koreans meeting the clinical criteria for AD ( n = 209), mild cognitive impairment ( n = 1449) or normal cognition ( n = 985). A missense variant, rs77359862 (R274W), in the SHANK-associated RH Domain Interactor ( SHARPIN ) gene was associated with entorhinal cortical thickness ( p = 5.0 × 10 −9 ) and hippocampal volume ( p = 5.1 × 10 −12 ). It revealed an increased risk of developing AD in the mediation analyses. This variant was also associated with amyloid-β accumulation ( p = 0.03) and measures of memory ( p = 1.0 × 10 −4 ) and executive function ( p = 0.04). We also found significant association of other SHARPIN variants with hippocampal volume in the Alzheimer’s Disease Neuroimaging Initiative (rs3417062, p = 4.1 × 10 −6 ) and AddNeuroMed (rs138412600, p = 5.9 × 10 −5 ) cohorts. Further, molecular dynamics simulations and co-immunoprecipitation indicated that the variant significantly reduced the binding of linear ubiquitination assembly complex proteins, SHPARIN and HOIL-1 Interacting Protein (HOIP), altering the downstream NF-κB signaling pathway. These findings suggest that SHARPIN plays an important role in the pathogenesis of AD.

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)0.000
metaresearch head score (Gemma)0.000
Version: codex-gemma-dda1882f352aValidation status: machine_predicted_unvalidated
Candidate categoriesInsufficient payload (model declined to judge)
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.580
Threshold uncertainty score0.999

Codex and Gemma teacher scores by category

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)0.0020.000

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 head0.049
GPT teacher head0.327
Teacher spread0.278 · 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.

Study designObservational
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

Citations20
Published2021
Admission routes1
Has abstractyes

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