Department of Molecular and Integrative Physiology, The University of Kansas Medical Center, Kansas City, KS, USA.
Department of Pathology and Laboratory Medicine, The University of Kansas Medical Center, Kansas City, KS, USA.
Cell Transplant. 2022 Jan-Dec;31:9636897221101116. doi: 10.1177/09636897221101116.
Kernicterus is a permanent condition caused by brain damage from bilirubin toxicity. Dystonia is one of the most debilitating symptoms of kernicterus and results from damage to the globus pallidus (GP). One potential therapeutic strategy to treat dystonia in kernicterus is to replace lost GP neurons and restore basal ganglia circuits through stem cell transplantation. Toward this end, we differentiated human embryonic stem cells (hESCs) into medial ganglion eminence (MGE; the embryological origin of most of the GP neurons)-like neural precursor cells (NPCs). We determined neurochemical phenotype in cell culture and after transplanting into the GP of jaundiced Gunn rats. We also determined grafted cell survival as well as migration, distribution, and morphology after transplantation. As in the GP, most cultured MGE-like NPCs expressed γ-aminobutyric acid (GABA), with some co-expressing markers for parvalbumin (PV) and others expressing markers for pro-enkephalin (PENK). MGE-like NPCs survived in brains at least 7 weeks after transplantation, with most aggregating near the injection site. Grafted cells expressed GABA and PV or PENK as in the normal GP. Although survival was low and the maturity of grafted cells varied, many cells produced neurite outgrowth. While promising, our results suggest the need to further optimize the differentiation protocol for MGE-like NPC for potential use in treating dystonia in kernicterus.
核黄疸是由胆红素毒性引起的脑损伤导致的永久性疾病。肌张力障碍是核黄疸最具致残性的症状之一,是由苍白球(GP)损伤引起的。通过干细胞移植来替代丢失的 GP 神经元并恢复基底神经节回路是治疗核黄疸肌张力障碍的一种潜在治疗策略。为此,我们将人胚胎干细胞(hESC)分化为中脑神经节隆起(MGE;大多数 GP 神经元的胚胎起源)样神经前体细胞(NPC)。我们在细胞培养中以及在黄疸 Gunn 大鼠的 GP 中移植后确定了神经化学表型。我们还确定了移植后的移植物存活以及迁移、分布和形态。与 GP 中的情况一样,大多数培养的 MGE 样 NPC 表达γ-氨基丁酸(GABA),有些同时表达副甲状腺球蛋白(PV)标志物,而另一些则表达前脑啡肽(PENK)标志物。MGE 样 NPC 在移植后至少 7 周内在大脑中存活,大多数聚集在注射部位附近。移植细胞表达 GABA 和 PV 或 PENK 与正常 GP 中的表达相同。尽管存活率较低且移植细胞的成熟度不同,但许多细胞产生了神经突生长。虽然有希望,但我们的结果表明需要进一步优化 MGE 样 NPC 的分化方案,以潜在用于治疗核黄疸的肌张力障碍。