Division of Hematology.
Blood. 2014 Jan 30;123(5):753-7. doi: 10.1182/blood-2013-10-530725. Epub 2013 Dec 13.
Megakaryocyte-specific transgene expression in patient-derived induced pluripotent stem cells (iPSCs) offers a new approach to study and potentially treat disorders affecting megakaryocytes and platelets. By using a Gp1ba promoter, we developed a strategy for achieving a high level of protein expression in human megakaryocytes. The feasibility of this approach was demonstrated in iPSCs derived from two patients with Glanzmann thrombasthenia (GT), an inherited platelet disorder caused by mutations in integrin αIIbβ3. Hemizygous insertion of Gp1ba promoter-driven human αIIb complementary DNA into the AAVS1 locus of iPSCs led to high αIIb messenger RNA and protein expression and correction of surface αIIbβ3 in megakaryocytes. Agonist stimulation of these cells displayed recovery of integrin αIIbβ3 activation. Our findings demonstrate a novel approach to studying human megakaryocyte biology as well as functional correction of the GT defect, offering a potential therapeutic strategy for patients with diseases that affect platelet function.
在患者来源的诱导多能干细胞 (iPSC) 中特异性表达巨核细胞可提供一种新方法来研究和潜在治疗影响巨核细胞和血小板的疾病。通过使用 Gp1ba 启动子,我们开发了一种在人巨核细胞中实现高水平蛋白表达的策略。该方法在源自两名 Glanzmann 血小板无力症 (GT) 患者的 iPSC 中得到了验证,GT 是一种由整合素 αIIbβ3 突变引起的遗传性血小板疾病。Gp1ba 启动子驱动的人 αIIb cDNA 的半合子插入到 iPSC 的 AAVS1 基因座中导致 αIIb mRNA 和蛋白表达升高,并纠正巨核细胞表面的 αIIbβ3。这些细胞的激动剂刺激显示整合素 αIIbβ3 激活的恢复。我们的发现证明了一种研究人类巨核细胞生物学的新方法,以及对 GT 缺陷的功能纠正,为影响血小板功能的疾病患者提供了一种潜在的治疗策略。