Draxin regulates interhemispheric fissure remodelling to influence the extent of corpus callosum formation

Laura Morcom, Timothy J. Edwards, Eric Rider, Dorothy Jones-Davis, Jonathan W.C. Lim, Kok Siong Chen, Ryan J. Dean, Jens Bunt, Yunan Ye, Ilan Gobius, Rodrigo Suárez, Simone Mandelstam, Elliott H. Sherr, Linda J. Richards

Onderzoeksoutput: Bijdrage aan tijdschriftArtikelpeer review

9 Citaten (Scopus)

Samenvatting

Corpus callosum dysgenesis (CCD) is a congenital disorder that incorporates either partial or complete absence of the largest cerebral commissure. Remodelling of the interhemispheric fissure (IHF) provides a substrate for callosal axons to cross between hemispheres, and its failure is the main cause of complete CCD. However, it is unclear whether defects in this process could give rise to the heterogeneity of expressivity and phenotypes seen in human cases of CCD. We identify incomplete IHF remodelling as the key structural correlate for the range of callosal abnormalities in inbred and outcrossed BTBR mouse strains, as well as in humans with partial CCD. We identify an eight base-pair deletion in Draxin and misregulated astroglial and leptomeningeal proliferation as genetic and cellular factors for variable IHF remodelling and CCD in BTBR strains. These findings support a model where genetic events determine corpus callosum structure by influencing leptomeningeal-astroglial interactions at the IHF.
Originele taal-2Engels
Artikelnummere61618
TijdschrifteLife
Volume10
DOI's
StatusGepubliceerd - 1 mei 2021
Extern gepubliceerdJa

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