Aguilar G Robert, Vidal Berta, Ji Hongzhu, Evenblij Joke, Liao Chien-Po, Ji Hongfei, Valperga Giulio, Fang-Yen Christopher, Hobert Oliver
Department of Biological Sciences, Howard Hughes Medical Institute, Columbia University, New York, New York, United States of America.
Technische Universität, Braunschweig, Germany.
PLoS Biol. 2025 Jan 6;23(1):e3002979. doi: 10.1371/journal.pbio.3002979. eCollection 2025 Jan.
Throughout the animal kingdom, several members of the basic helix-loop-helix (bHLH) family act as proneural genes during early steps of nervous system development. Roles of bHLH genes in specifying terminal differentiation of postmitotic neurons have been less extensively studied. We analyze here the function of 5 Caenorhabditis elegans bHLH genes, falling into 3 phylogenetically conserved subfamilies, which are continuously expressed in a very small number of postmitotic neurons in the central nervous system. We show (a) that 2 orthologs of the vertebrate bHLHe22/e23 genes, called hlh-17 and hlh-32, function redundantly to specify the identity of a single head interneuron class (AUA), as well as an individual motor neuron (VB2); (b) that the PTF1a ortholog hlh-13 acts as a terminal selector to control terminal differentiation and function of the sole octopaminergic neuron class in C. elegans, RIC; and (c) that the NHLH1/2 ortholog hlh-15 controls terminal differentiation and function of the peptidergic AVK head interneuron class, a known neuropeptidergic signaling hub in the animal. Strikingly, through null mutant analysis and cell-specific rescue experiments, we find that loss of hlh-15/NHLH in the peptidergic AVK neurons and the resulting abrogation of neuropeptide secretion from these neurons causes a substantially extended lifespan of the animal, which we propose to be akin to hypothalamic control of lifespan in vertebrates. Our functional analysis reveals themes of bHLH gene function during terminal differentiation that are complementary to the earlier lineage specification roles of other bHLH family members. However, such late functions are much more sparsely employed by members of the bHLH transcription factor family, compared to the function of the much more broadly employed homeodomain transcription factor family.
在整个动物界,碱性螺旋-环-螺旋(bHLH)家族的几个成员在神经系统发育的早期阶段作为神经源性基因发挥作用。bHLH基因在确定有丝分裂后神经元的终末分化方面的作用研究较少。我们在此分析了5个秀丽隐杆线虫bHLH基因的功能,它们分属于3个系统发育保守的亚家族,在中枢神经系统中极少数有丝分裂后神经元中持续表达。我们发现:(a)脊椎动物bHLHe22/e23基因的2个直系同源基因hlh-17和hlh-32,以冗余方式发挥作用,确定单个头部中间神经元类群(AUA)以及单个运动神经元(VB2)的身份;(b)PTF1a直系同源基因hlh-13作为终末选择因子,控制秀丽隐杆线虫唯一的章鱼胺能神经元类群RIC的终末分化和功能;(c)NHLH1/2直系同源基因hlh-15控制肽能AVK头部中间神经元类群的终末分化和功能,该类群是动物中已知的神经肽信号枢纽。引人注目的是,通过缺失突变分析和细胞特异性拯救实验,我们发现肽能AVK神经元中hlh-15/NHLH的缺失以及这些神经元神经肽分泌的消除导致动物寿命显著延长,我们认为这类似于脊椎动物下丘脑对寿命的控制。我们的功能分析揭示了bHLH基因在终末分化过程中的作用主题,这些主题与其他bHLH家族成员早期的谱系特化作用互补。然而,与广泛应用的同源域转录因子家族的功能相比,bHLH转录因子家族成员对这种后期功能的应用要少得多。