{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T22:07:31Z","timestamp":1787004451169,"version":"build-2736575974"},"reference-count":20,"publisher":"Portland Press Ltd.","issue":"10","license":[{"start":{"date-parts":[[2020,4,2]],"date-time":"2020-04-02T00:00:00Z","timestamp":1585785600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["portlandpress.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2020,5,29]]},"abstract":"<jats:p>Congenital dyserythropoietic anaemia (CDA) type I is a rare blood disorder characterised by moderate to severe macrocytic anaemia and hepatomegaly, with spongy heterochromatin and inter-nuclear bridges seen in bone marrow erythroblasts. The vast majority of cases of CDA type I are caused by mutations in the CDAN1 gene. The product of CDAN1 is Codanin-1, which interacts the histone chaperone ASF1 in the cytoplasm. Codanin-1 is a negative regulator of chromatin replication, sequestering ASF1 in the cytoplasm, restraining histone deposition and thereby limiting DNA replication. The remainder of CDA-I cases are caused by mutations in the C15ORF41 gene, but very little is known about the product of this gene. Here, we report that C15ORF41 forms a tight, near-stoichiometric complex with Codanin1 in human cells, interacting with the C-terminal region of Codanin-1. We present the characterisation of the C15ORF41\u2013Codanin-1 complex in humans in cells and in vitro, and demonstrate that Codanin-1 appears to sequester C15ORF41 in the cytoplasm as previously shown for ASF1. The findings in this study have major implications for understanding the functions of C15ORF41 and Codanin-1, and the aetiology of CDA-I.<\/jats:p>","DOI":"10.1042\/bcj20190944","type":"journal-article","created":{"date-parts":[[2020,4,2]],"date-time":"2020-04-02T06:38:04Z","timestamp":1585809484000},"page":"1893-1905","update-policy":"https:\/\/doi.org\/10.1042\/crossmark_policy","source":"Crossref","is-referenced-by-count":16,"title":["A complex comprising C15ORF41 and Codanin-1: the products of two genes mutated in congenital dyserythropoietic anaemia type I (CDA-I)"],"prefix":"10.1042","volume":"477","author":[{"given":"Maithili","family":"Shroff","sequence":"first","affiliation":[{"name":"MRC Protein Phosphorylation and Ubiquitylation Unit, School of Life Sciences, University of Dundee, Dundee DD1 5EH, U.K."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Axel","family":"Knebel","sequence":"additional","affiliation":[{"name":"MRC Protein Phosphorylation and Ubiquitylation Unit, School of Life Sciences, University of Dundee, Dundee DD1 5EH, U.K."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rachel","family":"Toth","sequence":"additional","affiliation":[{"name":"MRC Protein Phosphorylation and Ubiquitylation Unit, School of Life Sciences, University of Dundee, Dundee DD1 5EH, U.K."}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1183-8303","authenticated-orcid":false,"given":"John","family":"Rouse","sequence":"additional","affiliation":[{"name":"MRC Protein Phosphorylation and Ubiquitylation Unit, School of Life Sciences, University of Dundee, Dundee DD1 5EH, U.K."}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"288","published-online":{"date-parts":[[2020,5,28]]},"reference":[{"key":"2021112218074779300_BCJ-2019-0944C1","doi-asserted-by":"publisher","first-page":"178","DOI":"10.1016\/S0268-960X(98)90016-9","article-title":"Congenital dyserythropoietic anaemias: clinical features, haematological morphology and new biochemical data","volume":"12","year":"1998","journal-title":"Blood Rev."},{"key":"2021112218074779300_BCJ-2019-0944C2","doi-asserted-by":"publisher","first-page":"1034","DOI":"10.3324\/haematol.2009.014563","article-title":"The morphological diagnosis of congenital dyserythropoietic anemia: results of a quantitative analysis of peripheral blood and bone marrow cells","volume":"95","year":"2010","journal-title":"Haematologica"},{"key":"2021112218074779300_BCJ-2019-0944C3","first-page":"103","article-title":"Congenital dyserythropoietic anemia with karyorrhexis and multinuclearity of erythroblasts","volume":"34","year":"1968","journal-title":"Helv. 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