Jose Vicente Torres-Pérez, Sofia Anagianni, Eva Sheardown, Maria Elena Miletto-Petrazzini, Scott E Fraser, Brian Butterworth, Giorgio Vallortigara, Caroline H Brennan
{"title":"缺乏威廉姆斯综合征相关基因改变了斑马鱼的数量辨别。","authors":"Jose Vicente Torres-Pérez, Sofia Anagianni, Eva Sheardown, Maria Elena Miletto-Petrazzini, Scott E Fraser, Brian Butterworth, Giorgio Vallortigara, Caroline H Brennan","doi":"10.1016/j.bbr.2025.115860","DOIUrl":null,"url":null,"abstract":"<p><p>The ability to discriminate sets of items based on their numerosity is alleged to be an evolutionary conserved mechanism in all vertebrates. People with Williams syndrome (WS), a rare multigenic condition, show altered number and quantity cognition abilities. Assessing the contribution of specific genes to WS using animal models could help understand the basis of numerical impairments. Here, we assessed the quantitative abilities of juvenile zebrafish (Danio rerio) with loss of function of two of the genes affected in WS using a group size preference behavioural assay. The selected genes were: baz1b, implicated in neural crest development; and fzd9b, associated with neuronal functioning. The contrasts studied were 2 versus 5, 2 versus 4 and 2 versus 3. While group-level comparisons did not reveal statistically significant genotype differences, single-sample tests suggested a reduced preference for larger shoals in some contrasts among mutants. These trends were more apparent when the total number of items likely exceeded working memory capacity (i.e., 6 or more items), while performance on small numerosity contrasts remained relatively intact. These data agree with previous analyses of humans with WS and offer preliminary evidence that specific genes may influence quantity discrimination. Our research also supports the use of zebrafish as model organisms in which to characterise the neurobiological basis of dyscalculia in WS and associated disorders.</p>","PeriodicalId":8823,"journal":{"name":"Behavioural Brain Research","volume":" ","pages":"115860"},"PeriodicalIF":2.3000,"publicationDate":"2025-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Lack of Williams syndrome-associated genes alters quantity discrimination in zebrafish.\",\"authors\":\"Jose Vicente Torres-Pérez, Sofia Anagianni, Eva Sheardown, Maria Elena Miletto-Petrazzini, Scott E Fraser, Brian Butterworth, Giorgio Vallortigara, Caroline H Brennan\",\"doi\":\"10.1016/j.bbr.2025.115860\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>The ability to discriminate sets of items based on their numerosity is alleged to be an evolutionary conserved mechanism in all vertebrates. People with Williams syndrome (WS), a rare multigenic condition, show altered number and quantity cognition abilities. Assessing the contribution of specific genes to WS using animal models could help understand the basis of numerical impairments. Here, we assessed the quantitative abilities of juvenile zebrafish (Danio rerio) with loss of function of two of the genes affected in WS using a group size preference behavioural assay. The selected genes were: baz1b, implicated in neural crest development; and fzd9b, associated with neuronal functioning. The contrasts studied were 2 versus 5, 2 versus 4 and 2 versus 3. While group-level comparisons did not reveal statistically significant genotype differences, single-sample tests suggested a reduced preference for larger shoals in some contrasts among mutants. These trends were more apparent when the total number of items likely exceeded working memory capacity (i.e., 6 or more items), while performance on small numerosity contrasts remained relatively intact. These data agree with previous analyses of humans with WS and offer preliminary evidence that specific genes may influence quantity discrimination. Our research also supports the use of zebrafish as model organisms in which to characterise the neurobiological basis of dyscalculia in WS and associated disorders.</p>\",\"PeriodicalId\":8823,\"journal\":{\"name\":\"Behavioural Brain Research\",\"volume\":\" \",\"pages\":\"115860\"},\"PeriodicalIF\":2.3000,\"publicationDate\":\"2025-10-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Behavioural Brain Research\",\"FirstCategoryId\":\"102\",\"ListUrlMain\":\"https://doi.org/10.1016/j.bbr.2025.115860\",\"RegionNum\":3,\"RegionCategory\":\"心理学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q2\",\"JCRName\":\"BEHAVIORAL SCIENCES\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Behavioural Brain Research","FirstCategoryId":"102","ListUrlMain":"https://doi.org/10.1016/j.bbr.2025.115860","RegionNum":3,"RegionCategory":"心理学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q2","JCRName":"BEHAVIORAL SCIENCES","Score":null,"Total":0}
Lack of Williams syndrome-associated genes alters quantity discrimination in zebrafish.
The ability to discriminate sets of items based on their numerosity is alleged to be an evolutionary conserved mechanism in all vertebrates. People with Williams syndrome (WS), a rare multigenic condition, show altered number and quantity cognition abilities. Assessing the contribution of specific genes to WS using animal models could help understand the basis of numerical impairments. Here, we assessed the quantitative abilities of juvenile zebrafish (Danio rerio) with loss of function of two of the genes affected in WS using a group size preference behavioural assay. The selected genes were: baz1b, implicated in neural crest development; and fzd9b, associated with neuronal functioning. The contrasts studied were 2 versus 5, 2 versus 4 and 2 versus 3. While group-level comparisons did not reveal statistically significant genotype differences, single-sample tests suggested a reduced preference for larger shoals in some contrasts among mutants. These trends were more apparent when the total number of items likely exceeded working memory capacity (i.e., 6 or more items), while performance on small numerosity contrasts remained relatively intact. These data agree with previous analyses of humans with WS and offer preliminary evidence that specific genes may influence quantity discrimination. Our research also supports the use of zebrafish as model organisms in which to characterise the neurobiological basis of dyscalculia in WS and associated disorders.
期刊介绍:
Behavioural Brain Research is an international, interdisciplinary journal dedicated to the publication of articles in the field of behavioural neuroscience, broadly defined. Contributions from the entire range of disciplines that comprise the neurosciences, behavioural sciences or cognitive sciences are appropriate, as long as the goal is to delineate the neural mechanisms underlying behaviour. Thus, studies may range from neurophysiological, neuroanatomical, neurochemical or neuropharmacological analysis of brain-behaviour relations, including the use of molecular genetic or behavioural genetic approaches, to studies that involve the use of brain imaging techniques, to neuroethological studies. Reports of original research, of major methodological advances, or of novel conceptual approaches are all encouraged. The journal will also consider critical reviews on selected topics.