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Article

The non-canonical thioreductase Tmx2b is essential for neuronal survival during zebrafish embryonic brain development.

Development (Cambridge, England) - 15 Sept 2025

Dekker Jordy, Lam Wendy, van der Linde Herma C, Ophorst Floris, de Konink Charlotte, Schot Rachel, Kremers Gert-Jan, Sanderson Leslie E, Berdowski Woutje M, van Woerden Geeske M, Mancini Grazia M S, van Ham Tjakko J

Abstract excerpt

Biallelic variants in thioredoxin-related transmembrane 2 protein (TMX2) can cause a malformation of brain cortical development characterized by microcephaly, polymicrogyria and pachygyria by an unknown mechanism. To investigate and visualize how TMX2 loss disrupts brain development in vivo, we generated zebrafish deficient for TMX2 ortholog tmx2b, which during the first two developmental days showed normal brain...

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Identifiers

PMID: 40891441
DOI: 10.1242/dev.204348
PMCID: PMC12516324

Genes

TMX2

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