OtherAutism research : official journal of the International Society for Autism Research2025

Detailed phenotyping of Tbr1-2A-CreER knock-in mice demonstrates significant impacts on TBR1 protein levels and axon development.

Marissa Co, Grace K O'Brien, Kevin M Wright and 1 others

PMID 39548698

WHAT IT FOUND

The Tbr1-2A-CreER mouse line reduces TBR1 protein levels by 20.1% in heterozygotes and causes thinning of the anterior commissure.

Researchers advise caution when using this line for cortical projection neuron analysis due to these unintended developmental defects.

Key findings

01Heterozygous Tbr1-2A-CreER mice show a 20.1% reduction in TBR1 protein levels compared to wild-type controls.

02One copy of the Tbr1-2A-CreER allele causes thinning of the anterior commissure posterior limb in 7 of 7 mice examined.

03The authors advise caution when using this mouse line for analysis of cortical projection neurons because the knock-in creates a hypomorphic allele with altered protein levels.

STILL TO COME

How it was doneWhat they found

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What it does not show

The study was conducted in mice, not humans, so findings apply to experimental models rather than direct clinical practice. Molecular analyses were performed on bulk cortical tissue at a single early postnatal timepoint, which may miss cell-type-specific or later developmental effects. The authors did not rule out abnormalities in dendrite development, synapse formation, or synaptic function, which could contribute to the observed phenotypes.

Declared interests

The authors declare no competing financial interests.

The easy way to misread this

Do not assume this mouse line is a neutral tool for labeling neurons. The Tbr1-2A-CreER knock-in itself reduces TBR1 protein by 20.1% and alters brain development, which may confound results in autism-related research.

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The study

Certainty of evidence
Low

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    Marissa Co, Grace K O'Brien, Kevin M Wright, et al. Detailed phenotyping of Tbr1-2A-CreER knock-in mice demonstrates significant impacts on TBR1 protein levels and axon development. Autism research : official journal of the International Society for Autism Research. 2025.

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