Ohlemiller Kevin K, Kiener Anna L, Gagnon Patricia M
Department of Otolaryngology, Central Institute for the Deaf at Washington University School of Medicine, Fay and Carl Simons Center for Hearing and Deafness, Washington University School of Medicine, 660 S. Euclid, St. Louis, MO, 63110, USA.
Department of Speech and Hearing Science, Ohio State University, Columbus, OH, USA.
J Assoc Res Otolaryngol. 2016 Jun;17(3):173-94. doi: 10.1007/s10162-016-0558-8. Epub 2016 Mar 15.
We reported earlier that the endocochlear potential (EP) differs between C57BL/6J (B6) and BALB/cJ (BALB) mice, being lower in BALBs by about 10 mV (Ohlemiller et al. Hear Res 220: 10-26, 2006). This difference corresponds to strain differences with respect to the density of marginal cells in cochlear stria vascularis. After about 1 year of age, BALB mice also tend toward EP reduction that correlates with further marginal cell loss. We therefore suggested that early sub-clinical features of the BALB stria vascularis may predispose these mice to a condition modeling Schuknecht's strial presbycusis. We further reported (Ohlemiller et al. J Assoc Res Otolaryngol 12: 45-58, 2011) that the acute effects of a 2-h 110 dB SPL noise exposure differ between B6 and BALB mice, such that the EP remains unchanged in B6 mice, but is reduced by 40-50 mV in BALBs. In about 25 % of BALBs, the EP does not completely recover, so that permanent EP reduction may contribute to noise-induced permanent threshold shifts in BALBs. To identify genes and alleles that may promote natural EP variation as well as noise-related EP reduction in BALB mice, we have mapped related quantitative trait loci (QTLs) using 12 recombinant inbred (RI) strains formed from B6 and BALB (CxB1-CxB12). EP and strial marginal cell density were measured in B6 mice, BALB mice, their F1 hybrids, and RI mice without noise exposure, and 1-3 h after broadband noise (4-45 kHz, 110 dB SPL, 2 h). For unexposed mice, the strain distribution patterns for EP and marginal cell density were used to generate preliminary QTL maps for both EP and marginal cell density. Six QTL regions were at least statistically suggestive, including a significant QTL for marginal cell density on chromosome 12 that overlapped a weak QTL for EP variation. This region, termed Maced (Marginal cell density QTL) supports the notion of marginal cell density as a genetically influenced contributor to natural EP variation. Candidate genes for Maced notably include Foxg1, Foxa1, Akap6, Nkx2-1, and Pax9. Noise exposure produced significant EP reductions in two RI strains as well as significant EP increases in two RI strains. QTL mapping of the EP in noise-exposed RI mice yielded four suggestive regions. Two of these overlapped with QTL regions we previously identified for noise-related EP reduction in CBA/J mice (Ohlemiller et al. Hear Res 260: 47-53, 2010) on chromosomes 5 and 18 (Nirep). The present map may narrow the Nirep interval to a ~10-Mb region of proximal Chr. 18 that includes Zeb1, Arhgap12, Mpp7, and Gjd4. This study marks the first exploration of natural gene variants that modulate the EP. Their orthologs may underlie some human hearing loss that originates in the lateral wall.
我们之前报道过,内淋巴电位(EP)在C57BL/6J(B6)小鼠和BALB/cJ(BALB)小鼠之间存在差异,BALB小鼠的内淋巴电位比B6小鼠低约10 mV(Ohlemiller等人,《听觉研究》220: 10 - 26, 2006)。这种差异与耳蜗血管纹边缘细胞密度的品系差异相对应。大约1岁以后,BALB小鼠也倾向于内淋巴电位降低,这与边缘细胞的进一步丢失相关。因此,我们认为BALB血管纹的早期亚临床特征可能使这些小鼠易患模拟Schuknecht纹状老年性聋的病症。我们还进一步报道(Ohlemiller等人,《耳鼻咽喉头颈外科研究协会杂志》12: 45 - 58, 2011),2小时110 dB SPL噪声暴露的急性效应在B6小鼠和BALB小鼠之间有所不同,B6小鼠的内淋巴电位保持不变,而BALB小鼠的内淋巴电位降低40 - 50 mV。在大约25%的BALB小鼠中,内淋巴电位不能完全恢复,因此永久性内淋巴电位降低可能导致BALB小鼠出现噪声性永久性阈移。为了确定可能促进BALB小鼠自然内淋巴电位变化以及与噪声相关的内淋巴电位降低的基因和等位基因,我们使用由B6和BALB(CxB1 - CxB12)形成的12个重组近交(RI)品系绘制了相关数量性状位点(QTL)图谱。在未暴露于噪声的B6小鼠、BALB小鼠、它们的F1杂种小鼠和RI小鼠中,以及在宽带噪声(4 - 45 kHz,110 dB SPL,2小时)暴露后1 - 3小时测量内淋巴电位和血管纹边缘细胞密度。对于未暴露的小鼠,内淋巴电位和边缘细胞密度的品系分布模式用于生成内淋巴电位和边缘细胞密度的初步QTL图谱。六个QTL区域至少在统计学上具有提示意义,包括位于12号染色体上的一个边缘细胞密度显著QTL,它与一个内淋巴电位变化的弱QTL重叠。这个区域,称为Maced(边缘细胞密度QTL),支持边缘细胞密度是内淋巴电位自然变化的遗传影响因素这一观点。Maced的候选基因特别包括Foxg1、Foxa1、Akap6、Nkx2 - 1和Pax9。噪声暴露使两个RI品系的内淋巴电位显著降低,同时使两个RI品系的内淋巴电位显著升高。对噪声暴露的RI小鼠的内淋巴电位进行QTL定位产生了四个提示性区域。其中两个与我们之前在CBA/J小鼠中确定的与噪声相关的内淋巴电位降低的QTL区域(Ohlemiller等人,《听觉研究》260: 47 - 53, 2010)在5号和18号染色体(Nirep)上重叠。目前的图谱可能将Nirep区间缩小到18号染色体近端约10 Mb的区域,该区域包括Zeb1、Arhgap12、Mpp7和Gjd4。这项研究标志着对调节内淋巴电位的自然基因变体的首次探索。它们的直系同源基因可能是一些起源于外侧壁的人类听力损失的基础。