{"database":"biostudies-literature","file_versions":[],"scores":null,"additional":{"omics_type":["Unknown"],"volume":["56(1)"],"submitter":["Ogan BM"],"funding":["European Union and the Ministry of Education, Youth and Sports of the Czech Republic"],"pubmed_abstract":["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 FAM83H are known to cause amelogenesis imperfecta, highlighting its critical role in enamel formation. We generated Fam83h-deficient mice (Fam83h<sup>em2(IMPC)Ccpcz</sup>, Fam83h<sup>-/-</sup>) and mice lacking a part of the N-terminal CK1-binding domain (Fam83h<sup>∆87/∆87</sup>). Consistent with other Fam83h-deficient 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 t"],"journal":["European journal of immunology"],"pagination":["e70126"],"full_dataset_link":["https://www.ebi.ac.uk/biostudies/studies/S-EPMC12811513"],"repository":["biostudies-literature"],"pubmed_title":["FAM83H Regulates Postnatal T Cell Development Through Thymic Stroma Organization."],"pmcid":["PMC12811513"],"pubmed_authors":["Nickl P","Fedosieieva O","Balounova J","Krizova K","Prochazka J","Labaj J","Simova M","Tureckova J","Cervenkova S","Spoutil F","Prochazkova M","Dowling LJ","Vicikova K","Sedlacek R","Forstlova V","Ogan BM","Novosadova V"],"additional_accession":[]},"is_claimable":false,"name":"FAM83H Regulates Postnatal T Cell Development Through Thymic Stroma Organization.","description":"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 FAM83H are known to cause amelogenesis imperfecta, highlighting its critical role in enamel formation. We generated Fam83h-deficient mice (Fam83h<sup>em2(IMPC)Ccpcz</sup>, Fam83h<sup>-/-</sup>) and mice lacking a part of the N-terminal CK1-binding domain (Fam83h<sup>∆87/∆87</sup>). Consistent with other Fam83h-deficient 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 t","dates":{"release":"2026-01-01T00:00:00Z","publication":"2026 Jan","modification":"2026-07-15T06:34:57.969Z","creation":"2026-06-30T03:21:32.707Z"},"accession":"S-EPMC12811513","cross_references":{"pubmed":["41546142"],"doi":["10.1002/eji.70126"]}}