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DISC1-PML protein interaction for congenital CMV infection-induced cortical neural progenitor deficit: perturbance of host signaling via viral IE1

Saito, A.; Tankou, S.; Ishii, K.; Sakao-Suzuki, M.; Oh, E. C.; Murdoch, H.; Namkung, H.; Adelakun, S.; Furukori, K.; Fujimuro, M.; Salomoni, P.; Maul, G. G.; Hayward, G.; Tang, Q.; Yolken, R. H.; Houslay, M. D.; Katsanis, N.; Kosugi, I.; Yang, K.; Kamiya, A.; Ishizuka, K.; Sawa, A.

2025-09-07 developmental biology
10.1101/2025.09.03.674025 bioRxiv
Show abstract

Congenital CMV infection is the most common perinatal infection, affecting up to 0.5% of infants. This elicits long-term disabilities that include neuropsychiatric manifestations, such as intellectual disability, microcephaly. Despite its high prevalence, the underlying mechanism of how congenitally acquired CMV infection causes brain pathology remains unknown. Here we discovered the molecular interplay of key host (DISC1 and PML) and viral (IE1) proteins within the neural progenitor cells, which underlay an attenuated neural progenitor proliferation. Abolishing the viral IE1 protein by delivering IE1-targeting CRISPR/Cas9 to fetal brain rescued this progenitor cell deficit, a key pathology in congenital CMV infection. A selective targeting to a viral-specific protein by the CRISPR/Cas9 system is minimal in off-target effects. Therefore, we believe that a pivotal role of IE1 in an attenuated neural progenitor proliferation in the developing cortex through its interfering with interaction between host DISC1 and PML proteins.

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