Abstract
Riboflavin transporter 3 (RFVT3), encoded by the SLC52A3 gene, is important for riboflavin homeostasis in the small intestine, kidney, and placenta. Our previous study demonstrated that Slc52a3 knockout (Slc52a3−/−) mice exhibited neonatal lethality and metabolic disorder due to riboflavin deficiency. Here, we investigated the influence of Slc52a3 gene disruption on brain development using Slc52a3−/− embryos. Slc52a3−/− mice at postnatal day 0 showed hypoplasia of the brain and reduced thickness of cortical layers. At embryonic day 13.5, the formation of Tuj1+ neurons and Tbr2+ intermediate neural progenitors was significantly decreased; no significant difference was observed in the total number and proliferative rate of Pax6+ radial glia. Importantly, the hypoplastic phenotype was rescued upon riboflavin supplementation. Thus, it can be concluded that RFVT3 contributes to riboflavin homeostasis in embryos and that riboflavin itself is required during embryonic development of the cerebral cortex in mice.
Highlights
Riboflavin transporter 3 (RFVT3), encoded by the SLC52A3 gene, is important for riboflavin homeostasis in the small intestine, kidney, and placenta
We investigated the physiological role of RFVT3 using Slc52a3 knockout (Slc52a3−/−) mice
flavin mononucleotide (FMN), and flavin adenine dinucleotide (FAD) levels were significantly lower in Slc52a3−/− mice compared with WT animals (Fig. 1d–f)
Summary
Riboflavin transporter 3 (RFVT3), encoded by the SLC52A3 gene, is important for riboflavin homeostasis in the small intestine, kidney, and placenta. Human induced pluripotent stem cell-derived motor neurons from BVVL syndrome patients with SLC52A3 mutations were generated to explore the effect of riboflavin transporter mutations[28]. This in vitro disease model indicated a reduction in axonal growth and altered cytoskeletal functionality. The relationship between SLC52A3 gene disruption and neurological pathophysiology remains unclear.Here, we investigated the role of riboflavin in brain development using Slc52a3−/− mice
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