Fibrinogen inhibits sonic hedgehog signaling and impairs neonatal cerebellar development after blood-brain barrier disruption.
Olivia WeaverDawn GanoYungui ZhouHo Sung KimReshmi TognattaZhaoqi YanJae Kyu RyuCaroline BrandtTrisha BasuMartin GranaBelinda CabrigaMaria Del Pilar S AlzamoraAnthony J BarkovichKaterina AkassoglouMark A PetersenPublished in: Proceedings of the National Academy of Sciences of the United States of America (2024)
Cerebellar injury in preterm infants with central nervous system (CNS) hemorrhage results in lasting neurological deficits and an increased risk of autism. The impact of blood-induced pathways on cerebellar development remains largely unknown, so no specific treatments have been developed to counteract the harmful effects of blood after neurovascular damage in preterm infants. Here, we show that fibrinogen, a blood-clotting protein, plays a central role in impairing neonatal cerebellar development. Longitudinal MRI of preterm infants revealed that cerebellar bleeds were the most critical factor associated with poor cerebellar growth. Using inflammatory and hemorrhagic mouse models of neonatal cerebellar injury, we found that fibrinogen increased innate immune activation and impeded neurogenesis in the developing cerebellum. Fibrinogen inhibited sonic hedgehog (SHH) signaling, the main mitogenic pathway in cerebellar granule neuron progenitors (CGNPs), and was sufficient to disrupt cerebellar growth. Genetic fibrinogen depletion attenuated neuroinflammation, promoted CGNP proliferation, and preserved normal cerebellar development after neurovascular damage. Our findings suggest that fibrinogen alters the balance of SHH signaling in the neurovascular niche and may serve as a therapeutic target to mitigate developmental brain injury after CNS hemorrhage.
Keyphrases
- preterm infants
- blood brain barrier
- brain injury
- low birth weight
- oxidative stress
- traumatic brain injury
- magnetic resonance imaging
- mouse model
- gene expression
- innate immune
- signaling pathway
- computed tomography
- autism spectrum disorder
- dna methylation
- endothelial cells
- lps induced
- magnetic resonance
- inflammatory response
- cross sectional
- genome wide
- small molecule
- amino acid
- neural stem cells