{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,12]],"date-time":"2026-07-12T08:07:13Z","timestamp":1783843633233,"version":"3.55.0"},"reference-count":33,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2025,9,8]],"date-time":"2025-09-08T00:00:00Z","timestamp":1757289600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2025,9,8]],"date-time":"2025-09-08T00:00:00Z","timestamp":1757289600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100000050","name":"National Heart, Lung, and Blood Institute","doi-asserted-by":"publisher","award":["1R01HL168044"],"award-info":[{"award-number":["1R01HL168044"]}],"id":[{"id":"10.13039\/100000050","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["F31DE032881"],"award-info":[{"award-number":["F31DE032881"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000072","name":"National Institute of Dental and Craniofacial Research","doi-asserted-by":"publisher","award":["R01DE033009"],"award-info":[{"award-number":["R01DE033009"]}],"id":[{"id":"10.13039\/100000072","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100017618","name":"Division of Diabetes, Endocrinology, and Metabolic Diseases","doi-asserted-by":"publisher","award":["T32DK112751"],"award-info":[{"award-number":["T32DK112751"]}],"id":[{"id":"10.13039\/100017618","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000057","name":"National Institute of General Medical Sciences","doi-asserted-by":"publisher","award":["T32GM145441"],"award-info":[{"award-number":["T32GM145441"]}],"id":[{"id":"10.13039\/100000057","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["anatomypubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Developmental Dynamics"],"published-print":{"date-parts":[[2026,7]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      Gene transcription is crucial for embryo and postnatal development and is regulated by the Mediator complex. Mediator is comprised of four submodules, including the kinase submodule (CKM). The CKM consists of MED13, MED12, CDK8, and CCNC. In mammals, there are paralogs for CKM components, including MED13L, MED12L, and CDK19. Neurological disorders have been associated with mutations in CKM genes including\n                      <jats:italic>MED13L<\/jats:italic>\n                      syndrome.\n                      <jats:italic>MED13L<\/jats:italic>\n                      syndrome is generally characterized as a haploinsufficiency of MED13L with a broad phenotypic response due in part to a wide range of de novo mutations.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      We developed a\n                      <jats:italic>Med13l<\/jats:italic>\n                      heterozygous (HET) mouse model with an exon 11 deletion to evaluate whether\n                      <jats:italic>Med13l<\/jats:italic>\n                      HET mice are a viable research tool to study human phenotypes. We characterized our mouse model using growth, cardiovascular, and skeletal readouts. We observed\n                      <jats:italic>Med13l<\/jats:italic>\n                      HET mice are smaller than wildtype (WT) littermates, and over 60% of them exhibited one of two craniofacial anomalies: a pug snout with midface hypoplasia or a crooked snout. We also observed discontinuous squamosal sutures in a subset of our\n                      <jats:italic>Med13l<\/jats:italic>\n                      HETs.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      <jats:italic>Med13l<\/jats:italic>\n                      HET mice recapitulate\n                      <jats:italic>MED13L<\/jats:italic>\n                      syndrome phenotypes including a developmental growth delay and craniofacial anomalies.\n                      <jats:italic>Med13l<\/jats:italic>\n                      HET mice represent a novel research tool for\n                      <jats:italic>MED13L<\/jats:italic>\n                      syndrome.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/dvdy.70079","type":"journal-article","created":{"date-parts":[[2025,9,8]],"date-time":"2025-09-08T11:52:15Z","timestamp":1757332335000},"page":"724-735","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Heterozygous\n                    <i>Med13l<\/i>\n                    mice recapitulate a developmental growth delay and craniofacial anomalies seen in\n                    <scp>\n                      <i>MED13L<\/i>\n                    <\/scp>\n                    syndrome"],"prefix":"10.1002","volume":"255","author":[{"given":"Anna K.","family":"Leinheiser","sequence":"first","affiliation":[{"name":"Department of Internal Medicine, Division of Cardiovascular Medicine, Francois M. Abboud Cardiovascular Research Center, Fraternal Order of Eagles Diabetes Research Center University of Iowa  Iowa City Iowa USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Timothy T.","family":"Nguyen","sequence":"additional","affiliation":[{"name":"Interdisciplinary Graduate Program in Genetics University of Iowa  Iowa City Iowa USA"},{"name":"Iowa Institute for Oral Health Research, Department of Periodontics, College of Dentistry and Dental Clinics, Department of Anatomy and Cell Biology, Carver College of Medicine University of Iowa  Iowa City Iowa USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kayla M.","family":"Henry","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Division of Cardiovascular Medicine, Francois M. Abboud Cardiovascular Research Center, Fraternal Order of Eagles Diabetes Research Center University of Iowa  Iowa City Iowa USA"},{"name":"Interdisciplinary Graduate Program in Genetics University of Iowa  Iowa City Iowa USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mariela","family":"Rosales","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Division of Cardiovascular Medicine, Francois M. Abboud Cardiovascular Research Center, Fraternal Order of Eagles Diabetes Research Center University of Iowa  Iowa City Iowa USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eric","family":"Van Otterloo","sequence":"additional","affiliation":[{"name":"Interdisciplinary Graduate Program in Genetics University of Iowa  Iowa City Iowa USA"},{"name":"Iowa Institute for Oral Health Research, Department of Periodontics, College of Dentistry and Dental Clinics, Department of Anatomy and Cell Biology, Carver College of Medicine University of Iowa  Iowa City Iowa USA"},{"name":"Craniofacial Anomalies Research Center University of Iowa  Iowa City Iowa USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8950-742X","authenticated-orcid":false,"given":"Chad E.","family":"Grueter","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, Division of Cardiovascular Medicine, Francois M. Abboud Cardiovascular Research Center, Fraternal Order of Eagles Diabetes Research Center University of Iowa  Iowa City Iowa USA"},{"name":"Interdisciplinary Graduate Program in Genetics University of Iowa  Iowa City Iowa USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,9,8]]},"reference":[{"key":"e_1_2_9_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cell.2016.09.018"},{"key":"e_1_2_9_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41586-024-07069-w"},{"key":"e_1_2_9_4_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41576-023-00594-w"},{"key":"e_1_2_9_5_1","doi-asserted-by":"publisher","DOI":"10.1097\/WCO.0000000000000405"},{"key":"e_1_2_9_6_1","doi-asserted-by":"publisher","DOI":"10.1101\/cshperspect.a013839"},{"key":"e_1_2_9_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrg2901"},{"key":"e_1_2_9_8_1","doi-asserted-by":"publisher","DOI":"10.1038\/nrm3951"},{"key":"e_1_2_9_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.molcel.2004.05.006"},{"issue":"4","key":"e_1_2_9_10_1","article-title":"Mutual exclusivity of MED12\/MED12L, MED13\/13L, and CDK8\/19 paralogs revealed within the CDK\u2010mediator kinase 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