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Record W4402825491 · doi:10.1212/wnl.0000000000209832

Gene-Specific Effects on Brain Volume and Cognition of <i>TMEM106B</i> in Frontotemporal Lobar Degeneration

2024· article· en· W4402825491 on OpenAlexfundno aff
Marijne Vandebergh, Eliana Marisa Ramos, Nick Corriveau‐Lecavalier, Vijay K. Ramanan, John Kornak, Carly Mester, Tyler Kolander, Danielle Brushaber, Adam M. Staffaroni, Daniel H. Geschwind, Amy Wolf, Kejal Kantarci, Tania F. Gendron, Leonard Petrucelli, Marleen Van den Broeck, Sarah Wynants, Matthew Baker, Sergi Borrego‐Écija, Brian S. Appleby, Sami J. Barmada, Andrea Bozoki, David Clark, R. Ryan Darby, Bradford C. Dickerson, Kimiko Domoto‐Reilly, Julie A. Fields, Douglas Galasko, Nupur Ghoshal, Neill R. Graff‐Radford, Ian Grant, Lawrence S. Honig, Ging‐Yuek Robin Hsiung, Edward D. Huey, David J. Irwin, David S. Knopman, Justin Kwan, Gabriel C. Léger, Irene Litvan, Joseph C. Masdeu, Mario F. Mendez, Chiadi U. Onyike, Belén Pascual, Peter Pressman, Aaron Ritter, Erik D. Roberson, Allison Snyder, Anna Campbell Sullivan, Maria Carmela Tartaglia, Dylan Wint, Hilary W. Heuer, Leah K. Forsberg, Adam L. Boxer, Howard J. Rosen, Bradley F. Boeve, Rosa Rademakers

Bibliographic record

VenueNeurology · 2024
Typearticle
Languageen
FieldMedicine
TopicAmyotrophic Lateral Sclerosis Research
Canadian institutionsnot available
FundersNational Institute of Neurological Disorders and StrokeNational Institute of Mental HealthNational Institute on AgingCanadian Institutes of Health ResearchAvid RadiopharmaceuticalsAlzheimer SocietyParkinsonfondenUniversity of TorontoUniversity of California, San DiegoAlzheimer Society of B.C.ACADIA PharmaceuticalsGenentechEisaiUniversity of PennsylvaniaCurePSPUniversity of Southern CaliforniaPfizerBiogenEli Lilly and CompanyBrightFocus FoundationAstraZenecaBristol-Myers SquibbNational Institutes of HealthNational Ataxia FoundationTauRx PharmaceuticalsAlzheimer's Association
KeywordsFrontotemporal lobar degenerationFrontotemporal dementiaCognitionNeuroscienceDegeneration (medical)PsychologyMedicinePathologyDementiaDisease

Abstract

fetched live from OpenAlex

Background and Objectives TMEM106B has been proposed as a modifier of disease risk in FTLD-TDP, particularly in GRN pathogenic variant carriers. Furthermore, TMEM106B has been investigated as a disease modifier in the context of healthy aging and across multiple neurodegenerative diseases. The objective of this study was to evaluate and compare the effect of TMEM106B on gray matter volume and cognition in each of the common genetic FTD groups and in patients with sporadic FTD. Methods Participants were enrolled through the ARTFL/LEFFTDS Longitudinal Frontotemporal Lobar Degeneration (ALLFTD) study, which includes symptomatic and presymptomatic individuals with a pathogenic variant in C9orf72, GRN, MAPT, VCP, TBK1, TARDBP, symptomatic nonpathogenic variant carriers, and noncarrier family controls. All participants were genotyped for the TMEM106B rs1990622 SNP. Cross-sectionally, linear mixed-effects models were fitted to assess an association between TMEM106B and genetic group interaction with each outcome measure (gray matter volume and UDS3-EF for cognition), adjusting for education, age, sex, and CDR+NACC-FTLD sum of boxes. Subsequently, associations between TMEM106B and each outcome measure were investigated within the genetic group. For longitudinal modeling, linear mixed-effects models with time by TMEM106B predictor interactions were fitted. Results The minor allele of TMEM106B rs1990622, linked to a decreased risk of FTD, associated with greater gray matter volume in GRN pathogenic variant carriers under the recessive dosage model (N = 82, beta = 3.25, 95% CI [0.37–6.19], p = 0.034). This was most pronounced in the thalamus in the left hemisphere (beta = 0.03, 95% CI [0.01–0.06], p = 0.006), with a retained association when considering presymptomatic GRN pathogenic variant carriers only (N = 42, beta = 0.03, 95% CI [0.01–0.05], p = 0.003). The minor allele of TMEM106B rs1990622 also associated with greater cognitive scores among all C9orf72 pathogenic variant carriers (N = 229, beta = 0.36, 95% CI [0.05–0.066], p = 0.021) and in presymptomatic C9orf72 pathogenic variant carriers (N = 106, beta = 0.33, 95% CI [0.03–0.63], p = 0.036), under the recessive dosage model. Discussion We identified associations of TMEM106B with gray matter volume and cognition in the presence of GRN and C9orf72 pathogenic variants. The association of TMEM106B with outcomes of interest in presymptomatic GRN and C9orf72 pathogenic variant carriers could additionally reflect TMEM106B's effect on divergent pathophysiologic changes before the appearance of clinical symptoms.

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 machine prediction

Teacher imitation

Not calibrated prevalence, not ground truth. Human validation pending. The Gemma side is a direct model label for every work in the frame, read from the title-only record. The Codex side is a classifier learned from the 10,348 direct Codex labels and calibrated to design-weighted sample rates; fields without enough sample support carry no Codex call. Candidate is the union of the two sides; consensus is their intersection. These outputs are machine_predicted_unvalidated and are not human labels.

metaresearch head score (Codex)0.000
metaresearch head score (Gemma)0.001
Version: metacan-v3-hybrid-931329e0061cValidation status: machine_predicted_unvalidated
Candidate categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Observational · Consensus signal: Observational
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.003
Threshold uncertainty score0.008

Distilled classifier scores by category (both heads)

CategoryCodexGemma
Metaresearch0.0000.001
Meta-epidemiology (narrow)0.0000.000
Meta-epidemiology (broad)0.0000.001
Bibliometrics0.0010.000
Science and technology studies0.0000.000
Scholarly communication0.0000.000
Open science0.0000.000
Research integrity0.0010.000
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 head0.021
GPT teacher head0.276
Teacher spread0.255 · 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 source (direct Gemma or distilled Codex), not a consensus.

The models applied no category: nothing in the taxonomy fit this work.
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".

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Citations6
Published2024
Admission routes1
Has abstractyes

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