Annemarie Lange, Martin Bastmeyer, Joachim Bentrop
{"title":"遗传补偿强调了神经细胞粘附分子Ncam1在平衡斑马鱼侧线发育过程中的信号通路中的重要性","authors":"Annemarie Lange, Martin Bastmeyer, Joachim Bentrop","doi":"10.1016/j.ejcb.2025.151500","DOIUrl":null,"url":null,"abstract":"<div><div>The neural cell adhesion molecule NCAM1 is essential for neuronal development and enables organized cell migration, axon growth, and fasciculation. As a result of genome duplication in zebrafish, the paralogs Ncam1a and Ncam1b arose. Our previously published findings using morpholino knockdown experiments demonstrated the essential role of Ncam1b in the development of the zebrafish lateral line system, a mechanosensory organ critical for detecting water movements. <em>ncam1b</em> morphants exhibited severe defects, including impaired primordium migration, disrupted proneuromast deposition, and reduced cell proliferation within the primordium. These defects were linked to a disrupted interaction between Ncam1b and Fgfr1a, which led to compromised proliferation and abnormal lateral line development. The current study reveals that <em>ncam1b</em> mutants, however, unlike morphants, do not show this severe phenotype. Instead, we observed subtle alterations, including altered FGF and Wnt signaling and a redistribution of proliferating cells within the primordium. Notably, <em>ncam1b</em> mutants displayed elevated levels of the paralog <em>ncam1a</em> mRNA. The knockdown of either <em>ncam1a</em> or <em>upf3a</em> in <em>ncam1b</em> mutants resulted in a phenotype resembling that of <em>ncam1b</em> morphants. Upf3a is a key regulator of genetic compensation, a well-known phenomenon in zebrafish research. This supports the hypothesis that upregulated <em>ncam1a</em> compensates for the loss of <em>ncam1b</em>, facilitating normal lateral line development. These findings emphasize the essential role of Ncam1 in zebrafish lateral line development and suggest that the retention of both paralogs, <em>ncam1a</em> and <em>ncam1b</em>, acts as a protective mechanism to ensure the preservation of critical Ncam1 functions after gene duplication.</div></div>","PeriodicalId":12010,"journal":{"name":"European journal of cell biology","volume":"104 3","pages":"Article 151500"},"PeriodicalIF":4.3000,"publicationDate":"2025-06-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Genetic compensation highlights the importance of neural cell adhesion molecule Ncam1 paralogs in balancing signaling pathways during zebrafish lateral line development\",\"authors\":\"Annemarie Lange, Martin Bastmeyer, Joachim Bentrop\",\"doi\":\"10.1016/j.ejcb.2025.151500\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<div><div>The neural cell adhesion molecule NCAM1 is essential for neuronal development and enables organized cell migration, axon growth, and fasciculation. As a result of genome duplication in zebrafish, the paralogs Ncam1a and Ncam1b arose. Our previously published findings using morpholino knockdown experiments demonstrated the essential role of Ncam1b in the development of the zebrafish lateral line system, a mechanosensory organ critical for detecting water movements. <em>ncam1b</em> morphants exhibited severe defects, including impaired primordium migration, disrupted proneuromast deposition, and reduced cell proliferation within the primordium. These defects were linked to a disrupted interaction between Ncam1b and Fgfr1a, which led to compromised proliferation and abnormal lateral line development. The current study reveals that <em>ncam1b</em> mutants, however, unlike morphants, do not show this severe phenotype. Instead, we observed subtle alterations, including altered FGF and Wnt signaling and a redistribution of proliferating cells within the primordium. Notably, <em>ncam1b</em> mutants displayed elevated levels of the paralog <em>ncam1a</em> mRNA. The knockdown of either <em>ncam1a</em> or <em>upf3a</em> in <em>ncam1b</em> mutants resulted in a phenotype resembling that of <em>ncam1b</em> morphants. Upf3a is a key regulator of genetic compensation, a well-known phenomenon in zebrafish research. This supports the hypothesis that upregulated <em>ncam1a</em> compensates for the loss of <em>ncam1b</em>, facilitating normal lateral line development. These findings emphasize the essential role of Ncam1 in zebrafish lateral line development and suggest that the retention of both paralogs, <em>ncam1a</em> and <em>ncam1b</em>, acts as a protective mechanism to ensure the preservation of critical Ncam1 functions after gene duplication.</div></div>\",\"PeriodicalId\":12010,\"journal\":{\"name\":\"European journal of cell biology\",\"volume\":\"104 3\",\"pages\":\"Article 151500\"},\"PeriodicalIF\":4.3000,\"publicationDate\":\"2025-06-19\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"European journal of cell biology\",\"FirstCategoryId\":\"99\",\"ListUrlMain\":\"https://www.sciencedirect.com/science/article/pii/S0171933525000251\",\"RegionNum\":3,\"RegionCategory\":\"生物学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"CELL BIOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"European journal of cell biology","FirstCategoryId":"99","ListUrlMain":"https://www.sciencedirect.com/science/article/pii/S0171933525000251","RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"CELL BIOLOGY","Score":null,"Total":0}
Genetic compensation highlights the importance of neural cell adhesion molecule Ncam1 paralogs in balancing signaling pathways during zebrafish lateral line development
The neural cell adhesion molecule NCAM1 is essential for neuronal development and enables organized cell migration, axon growth, and fasciculation. As a result of genome duplication in zebrafish, the paralogs Ncam1a and Ncam1b arose. Our previously published findings using morpholino knockdown experiments demonstrated the essential role of Ncam1b in the development of the zebrafish lateral line system, a mechanosensory organ critical for detecting water movements. ncam1b morphants exhibited severe defects, including impaired primordium migration, disrupted proneuromast deposition, and reduced cell proliferation within the primordium. These defects were linked to a disrupted interaction between Ncam1b and Fgfr1a, which led to compromised proliferation and abnormal lateral line development. The current study reveals that ncam1b mutants, however, unlike morphants, do not show this severe phenotype. Instead, we observed subtle alterations, including altered FGF and Wnt signaling and a redistribution of proliferating cells within the primordium. Notably, ncam1b mutants displayed elevated levels of the paralog ncam1a mRNA. The knockdown of either ncam1a or upf3a in ncam1b mutants resulted in a phenotype resembling that of ncam1b morphants. Upf3a is a key regulator of genetic compensation, a well-known phenomenon in zebrafish research. This supports the hypothesis that upregulated ncam1a compensates for the loss of ncam1b, facilitating normal lateral line development. These findings emphasize the essential role of Ncam1 in zebrafish lateral line development and suggest that the retention of both paralogs, ncam1a and ncam1b, acts as a protective mechanism to ensure the preservation of critical Ncam1 functions after gene duplication.
期刊介绍:
The European Journal of Cell Biology, a journal of experimental cell investigation, publishes reviews, original articles and short communications on the structure, function and macromolecular organization of cells and cell components. Contributions focusing on cellular dynamics, motility and differentiation, particularly if related to cellular biochemistry, molecular biology, immunology, neurobiology, and developmental biology are encouraged. Manuscripts describing significant technical advances are also welcome. In addition, papers dealing with biomedical issues of general interest to cell biologists will be published. Contributions addressing cell biological problems in prokaryotes and plants are also welcome.