Department of Pediatrics, Affiliated Foshan Maternity & Child Healthcare Hospital, The Second School of Clinical Medicine of Southern Medical University, Foshan, China.
Hainan Women and Children's Medical Center, Haikou, China.
FEBS J. 2023 Jul;290(13):3369-3382. doi: 10.1111/febs.16761. Epub 2023 Mar 15.
Autism spectrum disorders (ASD) are associated with the contribution of many prenatal risk factors; in particular, the sex hormone progestin and vitamin D receptor (VDR) are associated with gastrointestinal (GI) symptoms in ASD development, although the related mechanism remains unclear. We investigated the possible role and mechanism of progestin 17-hydroxyprogesterone caproate (17-OHPC) exposure-induced GI dysfunction and autism-like behaviours (ALB) in mouse offspring. An intestine-specific VDR-deficient mouse model was established for prenatal treatment, while transplantation of haematopoietic stem cells (HSCT) with related gene manipulation was used for postnatal treatment for 17-OHPC exposure-induced GI dysfunction and ALB in mouse offspring. The in vivo mouse experiments found that VDR deficiency mimics prenatal 17-OHPC exposure-mediated GI dysfunction, but has no effect on 17-OHPC-mediated autism-like behaviours (ALB) in mouse offspring. Furthermore, prenatal 17-OHPC exposure induces CLDN1 suppression in intestine epithelial cells, and transplantation of HSCT with CLDN1 expression ameliorates prenatal 17-OHPC exposure-mediated GI dysfunction, but has no effect on 17-OHPC-mediated ALB in offspring. In conclusion, prenatal 17-OHPC exposure triggers GI dysfunction in autism-like mouse offspring via CLDN1 suppression, providing a possible explanation for the involvement of CLDN1 and VDR in prenatal 17-OHPC exposure-mediated GI dysfunction with ASD.
自闭症谱系障碍(ASD)与许多产前风险因素有关;特别是孕激素和维生素 D 受体(VDR)与 ASD 发展中的胃肠道(GI)症状有关,尽管相关机制尚不清楚。我们研究了孕激素 17-羟孕酮己酸酯(17-OHPC)暴露诱导的 GI 功能障碍和自闭症样行为(ALB)在小鼠后代中的可能作用和机制。建立了肠特异性 VDR 缺陷小鼠模型进行产前治疗,同时进行了造血干细胞(HSCT)移植及其相关基因操作的产后治疗,用于 17-OHPC 暴露诱导的 GI 功能障碍和自闭症样行为(ALB)。体内小鼠实验发现,VDR 缺陷模拟了产前 17-OHPC 暴露介导的 GI 功能障碍,但对 17-OHPC 介导的自闭症样行为(ALB)在小鼠后代中没有影响。此外,产前 17-OHPC 暴露诱导肠上皮细胞中 CLDN1 的抑制,而移植表达 CLDN1 的 HSCT 可改善产前 17-OHPC 暴露介导的 GI 功能障碍,但对后代中 17-OHPC 介导的自闭症样行为(ALB)没有影响。总之,产前 17-OHPC 暴露通过 CLDN1 的抑制引发自闭症样小鼠后代的 GI 功能障碍,为 CLDN1 和 VDR 参与产前 17-OHPC 暴露介导的 ASD 中的 GI 功能障碍提供了可能的解释。