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Record W2999911407 · doi:10.1002/jimd.12215

Magnetic resonance imaging reveals specific anatomical changes in the brain of Agat‐ and Gamt‐mice attributed to creatine depletion and guanidinoacetate alteration

2020· article· en· W2999911407 on OpenAlexaff
Ankit Sinha, Sohail Ahmed, Chris George, Melina Tsagaris, Amriya Naufer, Ingo von Both, Ilona Tkachyova, Matthijs van Eede, Mark Henkelman, Andreas Schulze

Bibliographic record

VenueJournal of Inherited Metabolic Disease · 2020
Typearticle
Languageen
FieldBiochemistry, Genetics and Molecular Biology
TopicMuscle metabolism and nutrition
Canadian institutionsUniversity of TorontoSickKids FoundationMental Health Research CanadaToronto Centre for PhenogenomicsHospital for Sick Children
Fundersnot available
KeywordsCreatineMagnetic resonance imagingNuclear magnetic resonanceNuclear magnetic resonance spectroscopyMedicineChemistryPathologyInternal medicineBiologyPhysicsRadiology

Abstract

fetched live from OpenAlex

Abstract Arginine:glycine amidinotransferase‐ and guanidinoacetate methyltransferase deficiency are severe neurodevelopmental disorders. It is not known whether mouse models of disease express a neuroanatomical phenotype. High‐resolution magnetic resonance imaging (MRI) with advanced image analysis was performed in perfused, fixed mouse brains encapsulated with the skull from male, 10‐12 week old Agat ‐exc and B6J.Cg‐Gamt tm1Isb mice (n = 48; n = 8 per genotype, strain). T2‐weighted MRI scans were nonlinearly aligned to a 3D atlas of the mouse brain with 62 structures identified. Local differences in brain shape related to genotype were assessed by analysis of deformation fields. Creatine (Cr) and guanidinoacetate (GAA) were measured with high‐performance liquid chromatography (HPLC) in brain homogenates (n = 24; n = 4 per genotype, strain) after whole‐body perfusion. Cr was decreased in the brain of Agat‐ and Gamt mutant mice. GAA was decreased in Agat −/− and increased in Gamt −/− . Body weight and brain volume were lower in Agat −/− than in Gamt −/− . The analysis of entire brain structures revealed corpus callosum, internal capsule, fimbria and hypothalamus being different between the genotypes in both strains. Eighteen and fourteen significant peaks (local areas of difference in relative size) were found in Agat‐ and Gamt mutants, respectively. Comparing Agat −/− with Gamt −/− , we found changes in three brain regions, lateral septum, amygdala, and medulla. Intra‐strain differences in four brain structures can be associated with Cr deficiency, while the inter‐strain differences in three brain structures of the mutant mice may relate to GAA. Correlating these neuroanatomical findings with gene expression data implies the role of Cr metabolism in the developing brain and the importance of early intervention in patients with Cr deficiency syndromes.

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 categoriesnone
Consensus categoriesnone
DomainCandidate signal: none · Consensus signal: none
Study designCandidate signal: Bench or experimental · Consensus signal: none
GenreCandidate signal: Empirical · Consensus signal: Empirical
Teacher disagreement score0.952
Threshold uncertainty score0.375

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)0.0000.000

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 head0.014
GPT teacher head0.249
Teacher spread0.234 · 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.

The models applied no category: nothing in the taxonomy fit this work.
Study designBench or experimental
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

Citations4
Published2020
Admission routes1
Has abstractyes

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