颅面和毛发发育过程中对 Alx4 功能的特异性要求

IF 2 3区 生物学 Q2 ANATOMY & MORPHOLOGY Developmental Dynamics Pub Date : 2024-03-13 DOI:10.1002/dvdy.705
Yu Lan, Zhaoming Wu, Han Liu, Rulang Jiang
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引用次数: 0

摘要

背景:ALX4 基因突变可导致常染色体显性顶骨乳突和常染色体隐性额叶发育不良伴脱发,但 ALX4 基因在颅面和其他发育过程中的作用机制尚不十分清楚。虽然以前曾报道过携带 Alx4 不同突变的小鼠,但同源突变体的围产期致死性以及 Alx4 在多种组织中的动态表达模式阻碍了系统地阐明 Alx4 在器官发生和疾病致病过程中的细胞和分子机制:结果:我们报告了Alx4f/f条件小鼠的产生,并表明分别在颅神经嵴和肢芽间充质中组织特异性Cre介导的Alx4失活再现了Alx4缺失小鼠的颅面和肢体发育缺陷,但不影响出生后存活。出生后存活的Alx4缺失小鼠表现出背侧脱发,而在神经嵴系中缺乏Alx4功能的小鼠表现出前颅骨高度受限的脱发区域,而在颅骨中胚层系中缺乏Alx4的小鼠则表现出正常的毛发发育,这表明Alx4在毛囊发育过程中在多个细胞系中发挥着部分冗余的作用:Alx4f/f小鼠为系统研究ALX家族转录因子在发育和疾病中的细胞类型和阶段特异性功能提供了宝贵的资源。
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Lineage-specific requirements of Alx4 function in craniofacial and hair development

Background

Disruption of ALX4 causes autosomal dominant parietal foramina and autosomal recessive frontonasal dysplasia with alopecia, but the mechanisms involving ALX4 in craniofacial and other developmental processes are not well understood. Although mice carrying distinct mutations in Alx4 have been previously reported, the perinatal lethality of homozygous mutants together with dynamic patterns of Alx4 expression in multiple tissues have hindered systematic elucidation of the cellular and molecular mechanisms involving Alx4 in organogenesis and disease pathogenesis.

Results

We report generation of Alx4f/f conditional mice and show that tissue-specific Cre-mediated inactivation of Alx4 in cranial neural crest and limb bud mesenchyme, respectively, recapitulated craniofacial and limb developmental defects as found in Alx4-null mice but without affecting postnatal survival. While Alx4-null mice that survive postnatally exhibited dorsal alopecia, mice lacking Alx4 function in the neural crest lineage exhibited a highly restricted region of hair loss over the anterior skull whereas mice lacking Alx4 in the cranial mesoderm lineage exhibited normal hair development, suggesting that Alx4 plays partly redundant roles in multiple cell lineages during hair follicle development.

Conclusion

The Alx4f/f mice provide a valuable resource for systematic investigation of cell type- and stage-specific function of ALX family transcription factors in development and disease.

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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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