Editorial highlights

IF 2 3区 生物学 Q2 ANATOMY & MORPHOLOGY
Paul A. Trainor
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Deficiencies in erythrocyte number or morphology, or hemoglobin levels can result in anemia. Zebrafish, which have transparent embryos, are a powerful model for studying human hematological disorders. In this study, the authors generated <i>epoa</i>-deficient zebrafish as a model of Diamond–Blackfan anemia like (DBAL), which occurs in humans in association with recessive loss-of-function mutations in EPO. EPO is crucial for erythrocyte development and oxygen transport and <i>epoa</i><sup><i>szy8/zy8</i></sup> mutants carrying the human EPO mutation c.530G&gt;A, developed DBAL due to reduced <i>EPO</i> expression. The severe anemia observed in <i>epoa</i><sup><i>szy8/zy8</i></sup> mutant zebrafish can be used to screen drugs for treating epoa-deficiency anemia, and recombinant human EPO significantly improved erythrocyte numbers. Zebrafish <i>epoa</i> models of DBAL are therefore beneficial for in vivo assessments of patient-derived <i>EPO</i> variants, and for developing potential therapeutic approaches for treating DBAL.</p><p><b>Craniofacial and Hair Development</b> “Lineage-specific requirements of Alx4 function in craniofacial and hair development” by Yu Lan, Zhaoming Wu, Han Liu, and Rulang Jiang; <i>Dev Dyn</i> 253:10, pp. 940–948. https://doi.org/10.1002/dvdy.705. The ALX family of transcription factors are key regulators of craniofacial development. Variants in <i>ALX4</i> have been associated with autosomal dominant parietal foramina and autosomal recessive frontonasal dysplasia with alopecia in humans, but the mechanisms connecting their etiology and pathogenesis remain poorly understood. <i>Alx4</i> is broadly expressed throughout development, making it difficult to determine its cell-autonomous and non-cell autonomous functions. Here the authors report the generation and characterization of <i>Alx4</i><sup><i>fx/fx</i></sup> conditional mice as a valuable new resource for investigating the pathogenic mechanisms underlying ALX4-related developmental disorders and alopecia. <i>Alx4</i> tissue-specific loss-of-function in neural crest cells and limb bud mesenchyme, results in craniofacial and limb bud developmental defects. <i>Alx4</i> null mutant mice that survive postnatally exhibit dorsal alopecia, whereas mice lacking <i>Alx4</i> in neural crest cells display restricted hair loss over the anterior skull. <i>Alx4</i> is expressed in mesenchymal cells surrounding the developing follicles, in outer root sheath epithelial cells surrounding the hair, and in the dermal papilla. Further study is therefore required to determine which domain of Alx4 is crucial for hair growth and regeneration.</p><p><b>Neural Development</b> “Reduced mTORC1-signaling in progenitor cells leads to retinal lamination deficits” by Christoffer Nord, Iwan Jones, Maria Garcia-Maestre, Anna-Carin Hägglund, and Leif Carlsson; <i>Dev Dyn</i> 253:10, pp. 922–939. https://doi.org/10.1002/dvdy.707. The mammalian central nervous system is remarkable for its anatomical complexity and functional capabilities. Underpinning these properties is neuronal lamination or the organization of distinct neuronal classes into stratified layers. For example, the mouse retina develops from a homogenous pool of retinal progenitor cells into six neuronal and one glial cell type whose cell bodies are laminated within three nuclear layers. Proper development of this tissue architecture requires coordinated cell proliferation, differentiation, and migration of progenitor cells. Here the authors demonstrate that the mammalian target of rapamycin complex 1 (mTORC1) signaling pathway is critical for the precise spatiotemporal regulation of retinal lamination mTOR is assembled from two core proteins (mTOR and Raptor) and associated regulators (PRAS40, mLST8, and Deptor). Tissue-specific ablation of Raptor in retinal progenitor cells resulted in decreased proliferation, increased apoptosis, irregular lamination and stratification, aberrant retinogeniculate topography, and loss of visually mediated behavior. Thus, mTORC1 regulates multiple aspects of retinal progenitor cell biology. This study therefore expands our understanding of the diverse roles of mTORC1 during visual system development and illustrates the conserved role of mTORC1 signaling in histogenesis of the CNS.</p>","PeriodicalId":11247,"journal":{"name":"Developmental Dynamics","volume":"253 10","pages":"880-881"},"PeriodicalIF":2.0000,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://onlinelibrary.wiley.com/doi/epdf/10.1002/dvdy.748","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Developmental Dynamics","FirstCategoryId":"99","ListUrlMain":"https://onlinelibrary.wiley.com/doi/10.1002/dvdy.748","RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"ANATOMY & MORPHOLOGY","Score":null,"Total":0}
引用次数: 0

Abstract

Every organism is a model organism for understanding development, evolution, disease, and regeneration, and we have only begun to scratch the surface of the interdisciplinary genetic, molecular, cellular, and developmental mechanisms that regulate these biological processes. These “Highlights” denote exciting advances recently reported in Developmental Dynamics that illustrate the complex dynamics of developmental biology.

Blood Development “Establishment of a Diamond-Blackfan anemia like (DBAL) model in zebrafish”, by Yiming Ling, Jiaye Wu, Yushi Liu, Panpan Meng, Ying Sun, Dejian Zhao, and Qing Lin; Dev Dyn 253:10, pp. 906–921. https://doi.org/10.1002/dvdy.703. Red blood cells (erythrocytes), which have a typical lifespan of 90–120 days, are essential for oxygen delivery throughout the body. Deficiencies in erythrocyte number or morphology, or hemoglobin levels can result in anemia. Zebrafish, which have transparent embryos, are a powerful model for studying human hematological disorders. In this study, the authors generated epoa-deficient zebrafish as a model of Diamond–Blackfan anemia like (DBAL), which occurs in humans in association with recessive loss-of-function mutations in EPO. EPO is crucial for erythrocyte development and oxygen transport and epoaszy8/zy8 mutants carrying the human EPO mutation c.530G>A, developed DBAL due to reduced EPO expression. The severe anemia observed in epoaszy8/zy8 mutant zebrafish can be used to screen drugs for treating epoa-deficiency anemia, and recombinant human EPO significantly improved erythrocyte numbers. Zebrafish epoa models of DBAL are therefore beneficial for in vivo assessments of patient-derived EPO variants, and for developing potential therapeutic approaches for treating DBAL.

Craniofacial and Hair Development “Lineage-specific requirements of Alx4 function in craniofacial and hair development” by Yu Lan, Zhaoming Wu, Han Liu, and Rulang Jiang; Dev Dyn 253:10, pp. 940–948. https://doi.org/10.1002/dvdy.705. The ALX family of transcription factors are key regulators of craniofacial development. Variants in ALX4 have been associated with autosomal dominant parietal foramina and autosomal recessive frontonasal dysplasia with alopecia in humans, but the mechanisms connecting their etiology and pathogenesis remain poorly understood. Alx4 is broadly expressed throughout development, making it difficult to determine its cell-autonomous and non-cell autonomous functions. Here the authors report the generation and characterization of Alx4fx/fx conditional mice as a valuable new resource for investigating the pathogenic mechanisms underlying ALX4-related developmental disorders and alopecia. Alx4 tissue-specific loss-of-function in neural crest cells and limb bud mesenchyme, results in craniofacial and limb bud developmental defects. Alx4 null mutant mice that survive postnatally exhibit dorsal alopecia, whereas mice lacking Alx4 in neural crest cells display restricted hair loss over the anterior skull. Alx4 is expressed in mesenchymal cells surrounding the developing follicles, in outer root sheath epithelial cells surrounding the hair, and in the dermal papilla. Further study is therefore required to determine which domain of Alx4 is crucial for hair growth and regeneration.

Neural Development “Reduced mTORC1-signaling in progenitor cells leads to retinal lamination deficits” by Christoffer Nord, Iwan Jones, Maria Garcia-Maestre, Anna-Carin Hägglund, and Leif Carlsson; Dev Dyn 253:10, pp. 922–939. https://doi.org/10.1002/dvdy.707. The mammalian central nervous system is remarkable for its anatomical complexity and functional capabilities. Underpinning these properties is neuronal lamination or the organization of distinct neuronal classes into stratified layers. For example, the mouse retina develops from a homogenous pool of retinal progenitor cells into six neuronal and one glial cell type whose cell bodies are laminated within three nuclear layers. Proper development of this tissue architecture requires coordinated cell proliferation, differentiation, and migration of progenitor cells. Here the authors demonstrate that the mammalian target of rapamycin complex 1 (mTORC1) signaling pathway is critical for the precise spatiotemporal regulation of retinal lamination mTOR is assembled from two core proteins (mTOR and Raptor) and associated regulators (PRAS40, mLST8, and Deptor). Tissue-specific ablation of Raptor in retinal progenitor cells resulted in decreased proliferation, increased apoptosis, irregular lamination and stratification, aberrant retinogeniculate topography, and loss of visually mediated behavior. Thus, mTORC1 regulates multiple aspects of retinal progenitor cell biology. This study therefore expands our understanding of the diverse roles of mTORC1 during visual system development and illustrates the conserved role of mTORC1 signaling in histogenesis of the CNS.

社论要点。
每种生物都是了解发育、进化、疾病和再生的模式生物,而我们对调控这些生物过程的跨学科遗传、分子、细胞和发育机制的研究才刚刚起步。这些 "亮点 "指出了《发育生物学》最近报道的令人振奋的进展,这些进展说明了发育生物学的复杂动态。Blood Development(血液发育):"Estabment of a Diamond-Blackfan anemia like (DBAL) model in zebrafish"(在斑马鱼中建立类似钻石-贝克范贫血症(DBAL)的模型),作者:Yiming Ling、Jiaye Wu、Yushi Liu、Panpan Meng、Ying Sun、Dejian Zhao 和 Qing Lin;Dev Dyn 253:10,第 906-921 页。https://doi.org/10.1002/dvdy.703。红细胞(红血球)的寿命一般为 90-120 天,是向全身输送氧气的重要物质。红细胞数量、形态或血红蛋白含量不足会导致贫血。斑马鱼的胚胎是透明的,是研究人类血液病的有力模型。在这项研究中,作者生成了epoa缺陷斑马鱼,作为类似钻石-贝克范贫血症(DBAL)的模型,这种贫血症发生在人类身上,与EPO的隐性功能缺失突变有关。EPO对红细胞发育和氧运输至关重要,携带人类EPO突变c.530G>A的epoaszy8/zy8突变体由于EPO表达减少而出现DBAL。在 epoaszy8/zy8 突变体斑马鱼中观察到的严重贫血症可用于筛选治疗环氧乙烷缺乏性贫血症的药物,重组人 EPO 可显著改善红细胞数量。因此,DBAL 的斑马鱼环氧乙烷模型有利于在体内评估源自患者的 EPO 变体,也有利于开发治疗 DBAL 的潜在疗法。颅面和毛发发育《Alx4 功能在颅面和毛发发育中的系谱特异性要求》,作者:Yu Lan、Zhaoming Wu、Han Liu 和 Rulang Jiang;Dev Dyn 253:10,第 940-948 页。https://doi.org/10.1002/dvdy.705。ALX 家族转录因子是颅面发育的关键调控因子。ALX4 的变异与人类常染色体显性顶骨乳突和常染色体隐性前额发育不良伴脱发有关,但其病因和发病机制仍不甚明了。Alx4 在整个发育过程中广泛表达,因此很难确定其细胞自主和非细胞自主功能。作者在本文中报告了Alx4fx/fx条件小鼠的产生和特征,这是研究ALX4相关发育障碍和脱发的致病机制的宝贵新资源。神经嵴细胞和肢芽间充质中的 Alx4 组织特异性功能缺失会导致颅面和肢芽发育缺陷。出生后存活下来的 Alx4 基因缺失突变小鼠表现出背侧脱发,而神经嵴细胞中缺乏 Alx4 的小鼠则表现出前颅骨局限性脱发。Alx4 在发育中的毛囊周围的间质细胞、毛发周围的外根鞘上皮细胞和真皮乳头中均有表达。Christoffer Nord、Iwan Jones、Maria Garcia-Maestre、Anna-Carin Hägglund和Leif Carlsson合著:《神经发育》(Dev Dyn 253:10,第922-939页),https://doi.org/10.1002/dvdy.707。哺乳动物的中枢神经系统因其解剖结构的复杂性和功能的强大而引人注目。这些特性的基础是神经元分层或将不同类别的神经元组织成分层。例如,小鼠视网膜从同质的视网膜祖细胞池发育成六种神经元和一种胶质细胞类型,其细胞体分层在三个核层中。这种组织结构的正常发育需要祖细胞协调的细胞增殖、分化和迁移。作者在本文中证明,哺乳动物雷帕霉素靶复合物 1(mTORC1)信号通路对于视网膜层叠的精确时空调控至关重要。组织特异性消减视网膜祖细胞中的 Raptor 会导致增殖减少、凋亡增加、不规则分层和分层、异常视网膜原形拓扑以及视觉介导行为的丧失。因此,mTORC1 调节视网膜祖细胞生物学的多个方面。因此,这项研究拓展了我们对 mTORC1 在视觉系统发育过程中的不同作用的理解,并说明了 mTORC1 信号在中枢神经系统组织发生过程中的保守作用。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
Developmental Dynamics
Developmental Dynamics 生物-发育生物学
CiteScore
5.10
自引率
8.00%
发文量
116
审稿时长
3-8 weeks
期刊介绍: Developmental Dynamics, is an official publication of the American Association for Anatomy. This peer reviewed journal provides an international forum for publishing novel discoveries, using any model system, that advances our understanding of development, morphology, form and function, evolution, disease, stem cells, repair and regeneration.
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