{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,3]],"date-time":"2026-06-03T00:10:30Z","timestamp":1780445430581,"version":"3.54.1"},"reference-count":47,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,1,16]],"date-time":"2026-01-16T00:00:00Z","timestamp":1768521600000},"content-version":"vor","delay-in-days":15,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Eur J Immunol"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Family of Sequence Similarity 83H (FAM83H\/SACK1H) is primarily expressed in epithelial cells, where it interacts with casein kinase 1 (CK1) and keratins to regulate cytoskeletal organization, cell proliferation, and vesicular trafficking. Mutations in\n                    <jats:italic>FAM83H<\/jats:italic>\n                    are known to cause amelogenesis imperfecta, highlighting its critical role in enamel formation. We generated\n                    <jats:italic>Fam83h<\/jats:italic>\n                    \u2010deficient mice (\n                    <jats:italic>\n                      Fam83h\n                      <jats:sup>em2(IMPC)Ccpcz<\/jats:sup>\n                      , Fam83h\n                      <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    <\/jats:italic>\n                    ) and mice lacking a part of the N\u2010terminal CK1\u2010binding domain (\n                    <jats:italic>\n                      Fam83h\n                      <jats:sup>\u220687\/\u220687<\/jats:sup>\n                    <\/jats:italic>\n                    ). Consistent with other\n                    <jats:italic>Fam83h<\/jats:italic>\n                    \u2010deficient models, these mice are subviable, smaller in size, and exhibit a sparse, scruffy coat, scaly skin, general weakness, and hypoactivity. Notably, both strains show impaired lymphoid cell development in early postnatal life. In the thymus,\n                    <jats:italic>Fam83h<\/jats:italic>\n                    expression is confined to thymic epithelial cells (TECs), and its deficiency in stromal cells results in disrupted thymic architecture and a severe block in the expansion of double\u2010negative stage 3 (DN3) T cells, ultimately leading to insufficient T cell production. Single\u2010cell transcriptomic analysis reveals that\n                    <jats:italic>\n                      Fam83h\n                      <jats:sup>\u2212\/\u2212<\/jats:sup>\n                    <\/jats:italic>\n                    cortical TECs (cTECs) express reduced levels of the TEC master regulator\n                    <jats:italic>Foxn1<\/jats:italic>\n                    , and its multiple downstream target genes, suggesting a critical role for FAM83H, likely in coordination with CK1, in cTEC maturation.\n                  <\/jats:p>","DOI":"10.1002\/eji.70126","type":"journal-article","created":{"date-parts":[[2026,1,17]],"date-time":"2026-01-17T04:50:58Z","timestamp":1768625458000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["FAM83H Regulates Postnatal T Cell Development Through Thymic Stroma Organization"],"prefix":"10.1002","volume":"56","author":[{"ORCID":"https:\/\/orcid.org\/0009-0001-8591-0841","authenticated-orcid":false,"given":"Betul Melike","family":"Ogan","sequence":"first","affiliation":[{"name":"Laboratory of Transgenic Models of Diseases Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"},{"name":"Department of Cell Biology Faculty of Science Charles University  Prague Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Veronika","family":"Forstlova","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura Jane","family":"Dowling","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michaela","family":"Simova","sequence":"additional","affiliation":[{"name":"Laboratory of Transgenic Models of Diseases Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"},{"name":"Department of Cell Biology Faculty of Science Charles University  Prague Czech Republic"},{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristina","family":"Vicikova","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vendula","family":"Novosadova","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frantisek","family":"Spoutil","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sylvie","family":"Cervenkova","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michaela","family":"Proch\u00e1zkov\u00e1","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jolana","family":"Tureckova","sequence":"additional","affiliation":[{"name":"Laboratory of Transgenic Models of Diseases Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olha","family":"Fedosieieva","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juraj","family":"Labaj","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Petr","family":"Nickl","sequence":"additional","affiliation":[{"name":"Laboratory of Transgenic Models of Diseases Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"},{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kamila","family":"Krizova","sequence":"additional","affiliation":[{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jan","family":"Prochazka","sequence":"additional","affiliation":[{"name":"Laboratory of Transgenic Models of Diseases Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"},{"name":"Czech Centre for Phenogenomics Institute of Molecular Genetics of the Czech Academy of Sciences  Vestec Czech Republic"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3352-392X","authenticated-orcid":false,"given":"Radislav","family":"Sedlacek","sequence":"additional","affiliation":[{"name":"Laboratory of Transgenic Models of Diseases Institute of Molecular Genetics of the Czech Academy 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