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A spatio-temporally constrained gene regulatory network directed by PBX1/2 acquires limb patterning specificity via HAND2 | bioRxiv

<!DOCTYPE html> <html lang="en" dir="ltr" xmlns="http://www.w3.org/1999/xhtml" xmlns:mml="http://www.w3.org/1998/Math/MathML"> <head prefix="og: http://ogp.me/ns# article: http://ogp.me/ns/article# book: http://ogp.me/ns/book#" > <!--[if IE]><![endif]--> <link rel="dns-prefetch" href="//d33xdlntwy0kbs.cloudfront.net" /> <link rel="dns-prefetch" href="//www.google.com" /> <link rel="dns-prefetch" href="//scholar.google.com" /> <link rel="dns-prefetch" href="//www.googletagmanager.com" /> <meta http-equiv="Content-Type" content="text/html; charset=utf-8" /> <link rel="shortcut icon" href="https://www.biorxiv.org/sites/default/files/images/favicon.ico" type="image/vnd.microsoft.icon" /> <meta name="viewport" content="width=device-width, initial-scale=1" /> <link rel="alternate" type="application/pdf" title="Full Text (PDF)" href="/content/10.1101/2022.03.08.483529v1.full.pdf" /> <link rel="alternate" type="text/plain" title="Full Text (Plain)" href="/content/10.1101/2022.03.08.483529v1.full.txt" /> <meta name="type" content="article" /> <meta name="category" content="article" /> <meta name="HW.identifier" content="/biorxiv/early/2022/03/09/2022.03.08.483529.atom" /> <meta name="HW.pisa" content="biorxiv;2022.03.08.483529v1" /> <meta name="DC.Format" content="text/html" /> <meta name="DC.Language" content="en" /> <meta name="DC.Title" content="A spatio-temporally constrained gene regulatory network directed by PBX1/2 acquires limb patterning specificity via HAND2" /> <meta name="DC.Identifier" content="10.1101/2022.03.08.483529" /> <meta name="DC.Date" content="2022-03-09" /> <meta name="DC.Publisher" content="Cold Spring Harbor Laboratory" /> <meta name="DC.Rights" content="© 2022, Posted by Cold Spring Harbor Laboratory. The copyright holder for this pre-print is the author. All rights reserved. The material may not be redistributed, re-used or adapted without the author's permission." /> <meta name="DC.AccessRights" content="restricted" /> <meta name="DC.Description" content="During development cell fates are specified by tightly controlled gene expression programs. PBX TALE transcription factors control gene regulatory networks (GRN) that direct vertebrate tissue patterning and organ morphogenesis. How PBX1/2 proteins acquire context-specific functions, despite widespread embryonic expression of Pbx1/2 , remains elusive. In mouse limb buds, mesenchymal-specific loss of PBX1/2 or of the transcriptional regulator HAND2 results in similar phenotypes, suggesting that PBX1/2- and HAND2-dependent programs converge to control limb development. To investigate this scenario, we combined tissue-specific and temporally-controlled mutagenesis with multi-omics approaches using the murine hindlimb bud as a model. We reconstructed a GRN collaboratively directed by PBX1/2 and HAND2, demonstrating that Pbx1 - Hand2 genetically interact in vivo during hindlimb patterning, with PBX1 concomitantly acting as an upstream regulator of Hand2 . At organismal-level resolution the GRN is active within restricted subsets of posterior-proximal hindlimb mesenchymal cells, wherein Pbx1/2 and Hand2 are co-expressed with their target genes. Genome-wide profiling of PBX1 binding across multiple tissues further revealed that HAND2 selects a subset of PBX-bound regions to impart limb patterning functionality. This research elucidates mechanisms underlying limb bud-specific functions by PBX1/2, while informing general principles by which promiscuous transcription factors cooperate with select cofactors to instruct distinct developmental programs. ### Competing Interest Statement The authors have declared no competing interest." /> <meta name="DC.Contributor" content="Marta Losa" /> <meta name="DC.Contributor" content="Iros Barozzi" /> <meta name="DC.Contributor" content="Marco Osterwalder" /> <meta name="DC.Contributor" content="Peyman Zarrineh" /> <meta name="DC.Contributor" content="Jean Denis Benazet" /> <meta name="DC.Contributor" content="Brandon Chacon" /> <meta name="DC.Contributor" content="Ausra Girdziusaite" /> <meta name="DC.Contributor" content="Angela Morabito" /> <meta name="DC.Contributor" content="Jianjian Zhu" /> <meta name="DC.Contributor" content="Susan Mackem" /> <meta name="DC.Contributor" content="Terence D. Capellini" /> <meta name="DC.Contributor" content="Nicoletta Bobola" /> <meta name="DC.Contributor" content="Diane Dickel" /> <meta name="DC.Contributor" content="Aimee Zuniga" /> <meta name="DC.Contributor" content="Axel Visel" /> <meta name="DC.Contributor" content="Rolf Zeller" /> <meta name="DC.Contributor" content="Licia Selleri" /> <meta name="article:published_time" content="2022-03-09" /> <meta name="article:section" content="New Results" /> <meta name="citation_title" content="A spatio-temporally constrained gene regulatory network directed by PBX1/2 acquires limb patterning specificity via HAND2" /> <meta name="citation_abstract" lang="en" content="&lt;h3&gt;ABSTRACT&lt;/h3&gt; &lt;p&gt;During development cell fates are specified by tightly controlled gene expression programs. PBX TALE transcription factors control gene regulatory networks (GRN) that direct vertebrate tissue patterning and organ morphogenesis. How PBX1/2 proteins acquire context-specific functions, despite widespread embryonic expression of &lt;i&gt;Pbx1/2&lt;/i&gt;, remains elusive. In mouse limb buds, mesenchymal-specific loss of PBX1/2 or of the transcriptional regulator HAND2 results in similar phenotypes, suggesting that PBX1/2- and HAND2-dependent programs converge to control limb development. To investigate this scenario, we combined tissue-specific and temporally-controlled mutagenesis with multi-omics approaches using the murine hindlimb bud as a model. We reconstructed a GRN collaboratively directed by PBX1/2 and HAND2, demonstrating that &lt;i&gt;Pbx1&lt;/i&gt;-&lt;i&gt;Hand2&lt;/i&gt; genetically interact &lt;i&gt;in vivo&lt;/i&gt; during hindlimb patterning, with PBX1 concomitantly acting as an upstream regulator of &lt;i&gt;Hand2&lt;/i&gt;. At organismal-level resolution the GRN is active within restricted subsets of posterior-proximal hindlimb mesenchymal cells, wherein &lt;i&gt;Pbx1/2&lt;/i&gt; and &lt;i&gt;Hand2&lt;/i&gt; are co-expressed with their target genes. Genome-wide profiling of PBX1 binding across multiple tissues further revealed that HAND2 selects a subset of PBX-bound regions to impart limb patterning functionality. This research elucidates mechanisms underlying limb bud-specific functions by PBX1/2, while informing general principles by which promiscuous transcription factors cooperate with select cofactors to instruct distinct developmental programs.&lt;/p&gt;" /> <meta name="citation_journal_title" content="bioRxiv" /> <meta name="citation_publisher" content="Cold Spring Harbor Laboratory" /> <meta name="citation_publication_date" content="2022/01/01" /> <meta name="citation_mjid" content="biorxiv;2022.03.08.483529v1" /> <meta name="citation_id" content="2022.03.08.483529v1" /> <meta name="citation_public_url" content="https://www.biorxiv.org/content/10.1101/2022.03.08.483529v1" /> <meta name="citation_abstract_html_url" content="https://www.biorxiv.org/content/10.1101/2022.03.08.483529v1.abstract" /> <meta name="citation_full_html_url" content="https://www.biorxiv.org/content/10.1101/2022.03.08.483529v1.full" /> <meta name="citation_pdf_url" content="https://www.biorxiv.org/content/biorxiv/early/2022/03/09/2022.03.08.483529.full.pdf" /> <meta name="citation_doi" content="10.1101/2022.03.08.483529" /> <meta name="citation_num_pages" content="42" /> <meta name="citation_article_type" content="Article" /> <meta name="citation_section" content="New Results" /> <meta name="citation_firstpage" content="2022.03.08.483529" /> <meta name="citation_author" content="Marta Losa" /> <meta name="citation_author_institution" content="Program in Craniofacial Biology, Institute for Human Genetics, Eli and Edythe Broad Center of Regeneration Medicine and Stem Cell Research, Department of Orofacial Sciences and Department of Anatomy, University of California at San Francisco" /> <meta name="citation_author" content="Iros Barozzi" /> <meta name="citation_author_institution" content="Center for Cancer Research, Medical University of Vienna" /> <meta name="citation_author" content="Marco Osterwalder" /> <meta name="citation_author_institution" content="Environmental Genomics and Systems Biology Division, Lawrence Berkeley National Laboratory" /> <meta name="citation_author_institution" content="Department for Biomedical Research (DBMR), University of Bern" /> <meta name="citation_author_institution" content="Department of Cardiology, Bern University Hospital" /> <meta name="citation_author_orcid" content="http://orcid.org/0000-0002-1969-2313" /> <meta name="citation_author" content="Peyman Zarrineh" /> <meta name="citation_author_institution" content="School of Medical Sciences, University of Manchester" /> <meta name="citation_author" content="Jean Denis Benazet" /> <meta name="citation_author_institution" content="Program in Craniofacial Biology, Institute for Human Genetics, Eli and Edythe Broad Center of Regeneration Medicine and Stem Cell Research, Department of Orofacial Sciences and Department of Anatomy, University of California at San Francisco" /> <meta name="citation_author" content="Brandon Chacon" /> <meta name="citation_author_institution" content="Program in Craniofacial Biology, Institute for Human Genetics, Eli and Edythe Broad Center of Regeneration Medicine and Stem Cell Research, Department of Orofacial Sciences and Department of Anatomy, University of California at San Francisco" /> <meta name="citation_author" content="Ausra Girdziusaite" /> <meta name="citation_author_institution" content="Developmental Genetics, Department Biomedicine, University of Basel" /> <meta name="citation_author" content="Angela Morabito" /> <meta name="citation_author_institution" content="Developmental Genetics, Department Biomedicine, University of Basel" /> <meta name="citation_author" content="Jianjian Zhu" /> <meta name="citation_author_institution" content="Cancer and Developmental Biology Laboratory, Center for Cancer Research, National Cancer Institute" /> <meta name="citation_author" content="Susan Mackem" /> <meta name="citation_author_institution" content="Cancer and Developmental Biology Laboratory, Center for Cancer Research, National Cancer Institute" /> <meta name="citation_author" content="Terence D. Capellini" /> <meta name="citation_author_institution" content="Department of Human Evolutionary Biology, Harvard University" /> <meta name="citation_author_institution" content="Broad Institute of Harvard and MIT" /> <meta name="citation_author" content="Nicoletta Bobola" /> <meta name="citation_author_institution" content="School of Medical Sciences, University of Manchester" /> <meta name="citation_author" content="Diane Dickel" /> <meta name="citation_author_institution" content="Environmental Genomics and Systems Biology Division, Lawrence Berkeley National Laboratory" /> <meta name="citation_author" content="Aimee Zuniga" /> <meta name="citation_author_institution" content="Developmental Genetics, Department Biomedicine, University of Basel" /> <meta name="citation_author_orcid" content="http://orcid.org/0000-0002-9953-3637" /> <meta name="citation_author" content="Axel Visel" /> <meta name="citation_author_institution" content="Environmental Genomics and Systems Biology Division, Lawrence Berkeley National Laboratory" /> <meta name="citation_author_institution" content="US Department of Energy Joint Genome Institute, Lawrence Berkeley National Laboratory" /> <meta name="citation_author_institution" content="School of Natural Sciences, University of California" /> <meta name="citation_author" content="Rolf Zeller" /> <meta name="citation_author_institution" content="Developmental Genetics, Department Biomedicine, University of Basel" /> <meta name="citation_author_orcid" content="http://orcid.org/0000-0002-3186-7403" /> <meta name="citation_author" content="Licia Selleri" /> <meta name="citation_author_institution" content="Program in Craniofacial Biology, Institute for Human Genetics, Eli and Edythe Broad Center of Regeneration Medicine and Stem Cell Research, Department of Orofacial Sciences and Department of Anatomy, University of California at San Francisco" /> <meta name="citation_author_email" content="licia.selleri@ucsf.edu" /> <meta name="citation_reference" content="Amândio AR, Beccari L, Lopez-Delisle L, Mascrez B, Zakany J, Gitto S, Duboule D. 2021. 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Current topics in developmental biology 139: 61–88. https://pubmed.ncbi.nlm.nih.gov/32450969/." /> <meta name="twitter:title" content="A spatio-temporally constrained gene regulatory network directed by PBX1/2 acquires limb patterning specificity via HAND2" /> <meta name="twitter:site" content="@biorxivpreprint" /> <meta name="twitter:card" content="summary" /> <meta name="twitter:image" content="https://www.biorxiv.org/sites/default/files/images/biorxiv_logo_homepage7-5-small.png" /> <meta name="twitter:description" content="During development cell fates are specified by tightly controlled gene expression programs. PBX TALE transcription factors control gene regulatory networks (GRN) that direct vertebrate tissue patterning and organ morphogenesis. How PBX1/2 proteins acquire context-specific functions, despite widespread embryonic expression of Pbx1/2 , remains elusive. In mouse limb buds, mesenchymal-specific loss of PBX1/2 or of the transcriptional regulator HAND2 results in similar phenotypes, suggesting that PBX1/2- and HAND2-dependent programs converge to control limb development. To investigate this scenario, we combined tissue-specific and temporally-controlled mutagenesis with multi-omics approaches using the murine hindlimb bud as a model. We reconstructed a GRN collaboratively directed by PBX1/2 and HAND2, demonstrating that Pbx1 - Hand2 genetically interact in vivo during hindlimb patterning, with PBX1 concomitantly acting as an upstream regulator of Hand2 . At organismal-level resolution the GRN is active within restricted subsets of posterior-proximal hindlimb mesenchymal cells, wherein Pbx1/2 and Hand2 are co-expressed with their target genes. Genome-wide profiling of PBX1 binding across multiple tissues further revealed that HAND2 selects a subset of PBX-bound regions to impart limb patterning functionality. This research elucidates mechanisms underlying limb bud-specific functions by PBX1/2, while informing general principles by which promiscuous transcription factors cooperate with select cofactors to instruct distinct developmental programs. ### Competing Interest Statement The authors have declared no competing interest." /> <meta name="og-title" property="og:title" content="A spatio-temporally constrained gene regulatory network directed by PBX1/2 acquires limb patterning specificity via HAND2" /> <meta name="og-url" property="og:url" content="https://www.biorxiv.org/content/10.1101/2022.03.08.483529v1" /> <meta name="og-site-name" property="og:site_name" content="bioRxiv" /> <meta name="og-description" property="og:description" content="During development cell fates are specified by tightly controlled gene expression programs. PBX TALE transcription factors control gene regulatory networks (GRN) that direct vertebrate tissue patterning and organ morphogenesis. How PBX1/2 proteins acquire context-specific functions, despite widespread embryonic expression of Pbx1/2 , remains elusive. In mouse limb buds, mesenchymal-specific loss of PBX1/2 or of the transcriptional regulator HAND2 results in similar phenotypes, suggesting that PBX1/2- and HAND2-dependent programs converge to control limb development. To investigate this scenario, we combined tissue-specific and temporally-controlled mutagenesis with multi-omics approaches using the murine hindlimb bud as a model. We reconstructed a GRN collaboratively directed by PBX1/2 and HAND2, demonstrating that Pbx1 - Hand2 genetically interact in vivo during hindlimb patterning, with PBX1 concomitantly acting as an upstream regulator of Hand2 . At organismal-level resolution the GRN is active within restricted subsets of posterior-proximal hindlimb mesenchymal cells, wherein Pbx1/2 and Hand2 are co-expressed with their target genes. Genome-wide profiling of PBX1 binding across multiple tissues further revealed that HAND2 selects a subset of PBX-bound regions to impart limb patterning functionality. This research elucidates mechanisms underlying limb bud-specific functions by PBX1/2, while informing general principles by which promiscuous transcription factors cooperate with select cofactors to instruct distinct developmental programs. ### Competing Interest Statement The authors have declared no competing interest." /> <meta name="og-type" property="og:type" content="article" /> <meta name="og-image" property="og:image" content="https://www.biorxiv.org/sites/default/files/images/biorxiv_logo_homepage7-5-small.png" /> <meta name="citation_date" content="2022-03-09" /> <link rel="alternate" type="application/vnd.ms-powerpoint" title="Powerpoint" href="/content/10.1101/2022.03.08.483529v1.ppt" /> <meta name="description" content="bioRxiv - the preprint server for biology, operated by Cold Spring Harbor Laboratory, a research and educational institution" /> <meta name="generator" content="Drupal 7 (http://drupal.org)" /> <link rel="canonical" 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data-apath="/biorxiv/early/2022/03/09/2022.03.08.483529.atom" data-hw-author-tooltip-instance="highwire_author_tooltip"><div class="highwire-cite highwire-cite-highwire-article highwire-citation-biorxiv-article-top clearfix has-author-tooltip" > <span class="biorxiv-article-type"> New Results </span> <h1 class="highwire-cite-title" id="page-title">A spatio-temporally constrained gene regulatory network directed by PBX1/2 acquires limb patterning specificity via HAND2</h1> <div class="highwire-cite-authors" ><span class="highwire-citation-authors"><span class="highwire-citation-author first" data-delta="0"><span class="nlm-given-names">Marta</span> <span class="nlm-surname">Losa</span></span>, <span class="highwire-citation-author" data-delta="1"><span class="nlm-given-names">Iros</span> <span class="nlm-surname">Barozzi</span></span>, <span class="highwire-citation-author hw-author-orcid-logo-wrapper" data-delta="2"><a href="http://orcid.org/0000-0002-1969-2313" target="_blank" class="hw-author-orcid-logo link-icon-only link-icon"><span class="hw-icon-orcid hw-icon-color-orcid"></span> <span class="title element-invisible">View ORCID Profile</span></a><span class="nlm-given-names">Marco</span> <span class="nlm-surname">Osterwalder</span></span>, <span class="highwire-citation-author" data-delta="3"><span class="nlm-given-names">Peyman</span> <span class="nlm-surname">Zarrineh</span></span>, <span class="highwire-citation-author" data-delta="4"><span class="nlm-given-names">Jean Denis</span> <span class="nlm-surname">Benazet</span></span>, <span class="highwire-citation-author" data-delta="5"><span class="nlm-given-names">Brandon</span> <span class="nlm-surname">Chacon</span></span>, <span class="highwire-citation-author" data-delta="6"><span class="nlm-given-names">Ausra</span> <span class="nlm-surname">Girdziusaite</span></span>, <span class="highwire-citation-author" data-delta="7"><span class="nlm-given-names">Angela</span> <span class="nlm-surname">Morabito</span></span>, <span class="highwire-citation-author" data-delta="8"><span class="nlm-given-names">Jianjian</span> <span class="nlm-surname">Zhu</span></span>, <span class="highwire-citation-author" data-delta="9"><span class="nlm-given-names">Susan</span> <span class="nlm-surname">Mackem</span></span>, <span class="highwire-citation-author" data-delta="10"><span class="nlm-given-names">Terence D.</span> <span class="nlm-surname">Capellini</span></span>, <span class="highwire-citation-author" data-delta="11"><span class="nlm-given-names">Nicoletta</span> <span class="nlm-surname">Bobola</span></span>, <span class="highwire-citation-author" data-delta="12"><span class="nlm-given-names">Diane</span> <span class="nlm-surname">Dickel</span></span>, <span class="highwire-citation-author hw-author-orcid-logo-wrapper" data-delta="13"><a href="http://orcid.org/0000-0002-9953-3637" target="_blank" class="hw-author-orcid-logo link-icon-only link-icon"><span class="hw-icon-orcid hw-icon-color-orcid"></span> <span class="title element-invisible">View ORCID Profile</span></a><span class="nlm-given-names">Aimee</span> <span class="nlm-surname">Zuniga</span></span>, <span class="highwire-citation-author" data-delta="14"><span class="nlm-given-names">Axel</span> <span class="nlm-surname">Visel</span></span>, <span class="highwire-citation-author hw-author-orcid-logo-wrapper" data-delta="15"><a href="http://orcid.org/0000-0002-3186-7403" target="_blank" class="hw-author-orcid-logo link-icon-only link-icon"><span class="hw-icon-orcid hw-icon-color-orcid"></span> <span class="title element-invisible">View ORCID Profile</span></a><span class="nlm-given-names">Rolf</span> <span class="nlm-surname">Zeller</span></span>, <span class="highwire-citation-author" data-delta="16"><span class="nlm-given-names">Licia</span> <span class="nlm-surname">Selleri</span></span></span></div> <div class="highwire-cite-metadata" ><span class="highwire-cite-metadata-doi highwire-cite-metadata"><span class="label">doi:</span> https://doi.org/10.1101/2022.03.08.483529 </span></div> </div> <div id="hw-article-author-popups-node-2437741--21842151150" style="display: none;"><div class="author-tooltip-0"><div class="author-tooltip-name">Marta Losa </div><div class="author-tooltip-affiliation"><span class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>^</span><span class='nlm-institution'>Program in Craniofacial Biology, Institute for Human Genetics, Eli and Edythe Broad Center of Regeneration Medicine and Stem Cell Research, Department of Orofacial Sciences and Department of Anatomy, University of California at San Francisco</span>, San Francisco, California, <span class='nlm-country'>USA</span></div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a 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class='nlm-sup'>#</span><span class='nlm-institution'>Environmental Genomics and Systems Biology Division, Lawrence Berkeley National Laboratory</span>, Berkeley, CA, <span class='nlm-country'>USA</span></div><div class='author-affiliation'><span class='nlm-sup'>x</span><span class='nlm-institution'>Department for Biomedical Research (DBMR), University of Bern</span>, Bern, <span class='nlm-country'>Switzerland</span></div><div class='author-affiliation'><span class='nlm-sup'>y</span><span class='nlm-institution'>Department of Cardiology, Bern University Hospital</span>, Bern, <span class='nlm-country'>Switzerland</span></div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Marco%2BOsterwalder%2B" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on Google Scholar</a></li><li class="author-tooltip-pubmed-link"><a 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class='nlm-institution'>Program in Craniofacial Biology, Institute for Human Genetics, Eli and Edythe Broad Center of Regeneration Medicine and Stem Cell Research, Department of Orofacial Sciences and Department of Anatomy, University of California at San Francisco</span>, San Francisco, California, <span class='nlm-country'>USA</span></div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Jean%2BDenis%2BBenazet%2B" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on Google Scholar</a></li><li class="author-tooltip-pubmed-link"><a href="/lookup/external-ref?access_num=Benazet%20JD&amp;link_type=AUTHORSEARCH" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on PubMed</a></li><li class="author-site-search-link last"><a href="/search/author1%3AJean%2BDenis%2BBenazet%2B" 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class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>#</span><span class='nlm-institution'>Environmental Genomics and Systems Biology Division, Lawrence Berkeley National Laboratory</span>, Berkeley, CA, <span class='nlm-country'>USA</span></div><div class='author-affiliation'><span class='nlm-sup'>()</span><span class='nlm-institution'>US Department of Energy Joint Genome Institute, Lawrence Berkeley National Laboratory</span>, Berkeley, CA 94720,<span class='nlm-country'>USA</span></div><div class='author-affiliation'><span class='nlm-sup'>z</span><span class='nlm-institution'>School of Natural Sciences, University of California</span>, Merced, Merced, CA 95343, <span class='nlm-country'>USA</span></div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Axel%2BVisel%2B" target="_blank" class="" data-icon-position="" 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class="author-tooltip-text"><div class='author-affiliation'><span class='nlm-sup'>^</span><span class='nlm-institution'>Program in Craniofacial Biology, Institute for Human Genetics, Eli and Edythe Broad Center of Regeneration Medicine and Stem Cell Research, Department of Orofacial Sciences and Department of Anatomy, University of California at San Francisco</span>, San Francisco, California, <span class='nlm-country'>USA</span></div></span></div><ul class="author-tooltip-find-more"><li class="author-tooltip-gs-link first"><a href="/lookup/google-scholar?link_type=googlescholar&amp;gs_type=author&amp;author%5B0%5D=Licia%2BSelleri%2B" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on Google Scholar</a></li><li class="author-tooltip-pubmed-link"><a href="/lookup/external-ref?access_num=Selleri%20L&amp;link_type=AUTHORSEARCH" target="_blank" class="" data-icon-position="" data-hide-link-title="0">Find this author on PubMed</a></li><li 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PBX TALE transcription factors control gene regulatory networks (GRN) that direct vertebrate tissue patterning and organ morphogenesis. How PBX1/2 proteins acquire context-specific functions, despite widespread embryonic expression of <em>Pbx1/2</em>, remains elusive. In mouse limb buds, mesenchymal-specific loss of PBX1/2 or of the transcriptional regulator HAND2 results in similar phenotypes, suggesting that PBX1/2- and HAND2-dependent programs converge to control limb development. To investigate this scenario, we combined tissue-specific and temporally-controlled mutagenesis with multi-omics approaches using the murine hindlimb bud as a model. We reconstructed a GRN collaboratively directed by PBX1/2 and HAND2, demonstrating that <em>Pbx1</em>-<em>Hand2</em> genetically interact <em>in vivo</em> during hindlimb patterning, with PBX1 concomitantly acting as an upstream regulator of <em>Hand2</em>. At organismal-level resolution the GRN is active within restricted subsets of posterior-proximal hindlimb mesenchymal cells, wherein <em>Pbx1/2</em> and <em>Hand2</em> are co-expressed with their target genes. Genome-wide profiling of PBX1 binding across multiple tissues further revealed that HAND2 selects a subset of PBX-bound regions to impart limb patterning functionality. This research elucidates mechanisms underlying limb bud-specific functions by PBX1/2, while informing general principles by which promiscuous transcription factors cooperate with select cofactors to instruct distinct developmental programs.</p></div><h3>Competing Interest Statement</h3><p id="p-5">The authors have declared no competing interest.</p><div class="section fn-group" id="fn-group-1"><h2>Footnotes</h2><ul><li class="fn-others" id="fn-2"><p id="p-2"><a class="rev-xref" href="#xref-fn-2-1">↵</a><span class="fn-label">[]</span> Co-senior authors</p></li></ul></div><span class="highwire-journal-article-marker-end"></span></div><span class="related-urls"></span></div></div> </div> </div> <div class="panel-separator"></div><div class="panel-pane pane-biorxiv-copyright" > <div class="pane-content"> <div class="field field-name-field-highwire-copyright field-type-text field-label-inline clearfix"><div class="field-label">Copyright&nbsp;</div><div class="field-items"><div class="field-item even">The copyright holder for this preprint is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity.<span class="license-type-none"> All rights reserved. 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class="outer collection depth-2 child"><div class = "data-wrapper"><a href="/collection/plant-biology" class="" data-icon-position="" data-hide-link-title="0">Plant Biology</a> <span class = "article-count">(11682)</span></div></li> <li class="outer collection depth-2 child"><div class = "data-wrapper"><a href="/collection/scientific-communication-and-education" class="" data-icon-position="" data-hide-link-title="0">Scientific Communication and Education</a> <span class = "article-count">(1780)</span></div></li> <li class="outer collection depth-2 child"><div class = "data-wrapper"><a href="/collection/synthetic-biology" class="" data-icon-position="" data-hide-link-title="0">Synthetic Biology</a> <span class = "article-count">(3272)</span></div></li> <li class="outer collection depth-2 child"><div class = "data-wrapper"><a href="/collection/systems-biology" class="" data-icon-position="" data-hide-link-title="0">Systems Biology</a> <span class = 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