Zegallai Hana M, Abu-El-Rub Ejlal, Olayinka-Adefemi Folayemi, Cole Laura K, Sparagna Genevieve C, Marshall Aaron J, Hatch Grant M
bioRxiv. 2021 Sep 8:2021.09.07.459330. doi: 10.1101/2021.09.07.459330.
Barth Syndrome (BTHS) is a rare X-linked genetic disorder caused by mutation in the TAFAZZIN gene which encodes the cardiolipin (CL) transacylase tafazzin (Taz). Taz deficiency in BTHS patients results in reduced CL in their tissues and a neutropenia which contributes to the risk of infections. However, the impact of Taz deficiency in other cells of the immune system is poorly understood. Mesenchymal stem cells (MSCs) are well known for their immune inhibitory function. We examined whether Taz-deficiency in murine MSCs impacted their ability to modulate lipopolysaccharide (LPS)-activated wild type (WT) murine B lymphocytes. MSCs from tafazzin knockdown (TazKD) mice exhibited a 50% reduction in CL compared to wild type (WT) MSCs. However, mitochondrial oxygen consumption rate and membrane potential were unaltered. In contrast, TazKD MSCs exhibited increased glycolysis compared to WT MSCs and this was associated with elevated proliferation, phosphatidylinositol-3-kinase expression and expression of the immunosuppressive markers indoleamine-2,3-dioxygenase, cytotoxic T-lymphocyte-associated protein 4, interleukin-10, and cluster of differentiation 59. When co-cultured with LPS-activated WT B cells, TazKD MSCs inhibited B cell proliferation and growth rate and reduced B cell secretion of IgM to a greater extent than B cells co-cultured with WT MSCs. In addition, co-culture of LPS-activated WT B cells with TazKD MSCs induced B cell differentiation toward potent immunosuppressive phenotypes including interleukin-10 secreting plasma cells and B regulatory cells compared to activated B cells co-cultured with WT MSCs. These results indicate that Taz deficiency in MSCs enhances MSCs-mediated immunosuppression of activated B lymphocytes.
巴氏综合征(BTHS)是一种罕见的X连锁遗传病,由编码心磷脂(CL)转酰基酶塔法兹蛋白(Taz)的TAFAZZIN基因突变引起。BTHS患者体内Taz缺乏导致组织中CL减少以及中性粒细胞减少,从而增加感染风险。然而,Taz缺乏对免疫系统其他细胞的影响尚不清楚。间充质干细胞(MSC)以其免疫抑制功能而闻名。我们研究了小鼠MSC中Taz缺乏是否会影响其调节脂多糖(LPS)激活的野生型(WT)小鼠B淋巴细胞的能力。与野生型(WT)MSC相比,来自塔法兹蛋白敲低(TazKD)小鼠的MSC的CL减少了50%。然而,线粒体氧消耗率和膜电位未发生改变。相反, 与WT MSC相比,TazKD MSC的糖酵解增加,这与增殖增加、磷脂酰肌醇-3-激酶表达以及免疫抑制标志物吲哚胺-2,3-双加氧酶、细胞毒性T淋巴细胞相关蛋白4、白细胞介素-10和分化簇59的表达升高有关。当与LPS激活的WT B细胞共培养时,TazKD MSC比与WT MSC共培养的B细胞更能抑制B细胞增殖和生长速率,并更大程度地降低B细胞分泌IgM。此外,与WT MSC共培养的激活B细胞相比,LPS激活的WT B细胞与TazKD MSC共培养可诱导B细胞分化为强效免疫抑制表型,包括分泌白细胞介素-10的浆细胞和B调节细胞。这些结果表明,MSC中Taz缺乏增强了MSC介导的对激活B淋巴细胞的免疫抑制作用。