Program in Developmental Biology, Baylor College of Medicine, Houston, TX, USA.
PLoS One. 2013;8(4):e60450. doi: 10.1371/journal.pone.0060450. Epub 2013 Apr 2.
Male Drosophila are monosomic for the X chromosome, but survive due to dosage compensation. They use the Male Specific Lethal (MSL) complex composed of noncoding roX RNA and histone modifying enzymes to hypertranscribe most genes along the X ∼1.6-1.8 fold relative to each female allele. It is not known how the MSL complex achieves this precise adjustment to a large and diverse set of target genes. We carried out a genetic screen searching for novel factors that regulate dosage compensation in flies. This strategy generated thirty alleles in a previously uncharacterized gene, over compensating males (ocm) that antagonizes some aspect of MSL activity. The mutations were initially recovered because they derepressed an MSL-dependent eye color reporter. Null ocm mutations are lethal to both sexes early in development revealing an essential function. Combinations of hypomorphic ocm alleles display a male specific lethality similar to mutations in the classic msl genes, but ocm males die due to excessive, rather than lack of dosage compensation. Males that die due to very low MSL activity can be partially rescued by ocm mutations. Likewise, males that would die from ocm mutations can be rescued by reducing the dose of various msl and roX genes. ocm encodes a large nuclear protein that shares a novel cysteine rich motif with known transcription factors.
雄性果蝇的 X 染色体是单体的,但由于剂量补偿而存活。它们使用由非编码 roX RNA 和组蛋白修饰酶组成的雄性特异致死 (MSL) 复合物,使大多数基因的转录水平相对于每个雌性等位基因增加 1.6-1.8 倍。目前尚不清楚 MSL 复合物如何对大量不同的靶基因进行这种精确的调整。我们进行了一项遗传筛选,以寻找调节果蝇剂量补偿的新因子。该策略在一个以前未被描述的基因中产生了三十个等位基因,这些等位基因过度补偿雄性(ocm),拮抗 MSL 活性的某些方面。最初恢复这些突变是因为它们解除了 MSL 依赖性眼色报告基因的抑制。ocm 缺失突变在发育早期对雌雄两性都是致命的,这揭示了其必需的功能。功能减弱的 ocm 等位基因的组合表现出类似于经典 msl 基因的雄性特异性致死,但 ocm 雄性由于过度而不是缺乏剂量补偿而死亡。由于 MSL 活性非常低而死亡的雄性可以部分被 ocm 突变拯救。同样,由于 ocm 突变而死亡的雄性可以通过降低各种 msl 和 roX 基因的剂量来拯救。ocm 编码一种大型核蛋白,它与已知的转录因子共享一个新的富含半胱氨酸的基序。