{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,20]],"date-time":"2026-02-20T08:36:39Z","timestamp":1771576599195,"version":"3.50.1"},"reference-count":45,"publisher":"The Company of Biologists","issue":"4","license":[{"start":{"date-parts":[[2025,4,28]],"date-time":"2025-04-28T00:00:00Z","timestamp":1745798400000},"content-version":"vor","delay-in-days":27,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100024731","name":"AABB Foundation","doi-asserted-by":"crossref","award":["NBF 22-09R"],"award-info":[{"award-number":["NBF 22-09R"]}],"id":[{"id":"10.13039\/100024731","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000968","name":"American Heart Association","doi-asserted-by":"crossref","award":["23CDA1053102"],"award-info":[{"award-number":["23CDA1053102"]}],"id":[{"id":"10.13039\/100000968","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000062","name":"National Institute of Diabetes and Digestive and Kidney Diseases","doi-asserted-by":"crossref","award":["U24DK126127"],"award-info":[{"award-number":["U24DK126127"]}],"id":[{"id":"10.13039\/100000062","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Kansas Institute for Precision Medicine Center of Biomedical Research Excellence","award":["COBRE-P20 GM130423"],"award-info":[{"award-number":["COBRE-P20 GM130423"]}]},{"DOI":"10.13039\/501100010462","name":"National Institute of Genetics","doi-asserted-by":"crossref","award":["18A2023"],"award-info":[{"award-number":["18A2023"]}],"id":[{"id":"10.13039\/501100010462","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01HL157975-01A1"],"award-info":[{"award-number":["R01HL157975-01A1"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"crossref","award":["R01HL164016-01A1"],"award-info":[{"award-number":["R01HL164016-01A1"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"crossref"}]},{"name":"University of Kansas Medical Center"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,4,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Mutations in the 5\u2032-untranslated region (5\u2032-UTR) of ankyrin repeat domain-containing protein 26 (ANKRD26) are associated with hereditary thrombocytopenia 2 (THC2). However, the causative role of these mutations and the mechanisms underlying THC2 are not fully established. Here, we report, for the first time, that zebrafish carrying a deletion of two nucleotides (\u03942) in the 5\u2032-UTR of ankrd26 recapitulate some of the key laboratory features of THC2. ankrd26ku6 (homozygous for the \u03942 deletion in the 5\u2032-UTR) fish larvae exhibited significantly increased expression of ankrd26 mRNA and protein. Adult ankrd26ku6 fish exhibited spontaneous thrombocytopenia. Furthermore, the thrombocytes from ankrd26ku6 fish showed enhanced ability to adhere and aggregate on a collagen surface under flow. Proteomic profiling demonstrated marked upregulation of Ninjurin 1 in young thrombocytes from ankrd26ku6 fish compared with those from wild-type controls. The ankrd26ku6 fish with a homozygous nacre allele developed myelodysplastic syndrome at old age. ANKRD26 protein levels were also significantly increased in platelets and plasma from patients with immune thrombotic thrombocytopenic purpura compared with those from unaffected controls. We conclude that ANKRD26 overexpression, resulting from either hereditary or acquired mechanisms, contributes to thrombocytopenia, thrombosis and hematologic malignancies.<\/jats:p>","DOI":"10.1242\/dmm.052222","type":"journal-article","created":{"date-parts":[[2025,4,2]],"date-time":"2025-04-02T06:46:08Z","timestamp":1743576368000},"source":"Crossref","is-referenced-by-count":2,"title":["Modeling <i>ANKRD26<\/i> 5\u2032-UTR mutation-related thrombocytopenia"],"prefix":"10.1242","volume":"18","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0120-9343","authenticated-orcid":false,"given":"Liang","family":"Zheng","sequence":"first","affiliation":[{"name":"The University of Kansas Medical Center 1 Department of Pathology and Laboratory Medicine , , Kansas City, KS 66126 ,","place":["USA"]},{"name":"Institute of Reproductive and Developmental Sciences, The University of Kansas Medical Center 2 , Kansas City, KS 66126 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Zhijian","family":"Wu","sequence":"additional","affiliation":[{"name":"The University of Kansas Medical Center 1 Department of Pathology and Laboratory Medicine , , Kansas City, KS 66126 ,","place":["USA"]},{"name":"The Second Xiangya Hospital, Central South University 3 Department of Pulmonary and Critical Care Medicine , , Changsha 410010 ,","place":["China"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Noritaka","family":"Yada","sequence":"additional","affiliation":[{"name":"The University of Kansas Medical Center 1 Department of Pathology and Laboratory Medicine , , Kansas City, KS 66126 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Szumam","family":"Liu","sequence":"additional","affiliation":[{"name":"The University of Kansas Medical Center 1 Department of Pathology and Laboratory Medicine , , Kansas City, KS 66126 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Cindy","family":"Lin","sequence":"additional","affiliation":[{"name":"The University of Kansas Medical Center 1 Department of Pathology and Laboratory Medicine , , Kansas City, KS 66126 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Antonia","family":"Bignotti","sequence":"additional","affiliation":[{"name":"The University of Kansas Medical Center 1 Department of Pathology and Laboratory Medicine , , Kansas City, KS 66126 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Xinyang","family":"Zhao","sequence":"additional","affiliation":[{"name":"The University of Kansas Medical Center 1 Department of Pathology and Laboratory Medicine , , Kansas City, KS 66126 ,","place":["USA"]},{"name":"Institute of Reproductive and Developmental Sciences, The University of Kansas Medical Center 2 , Kansas City, KS 66126 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1680-5295","authenticated-orcid":false,"given":"X. Long","family":"Zheng","sequence":"additional","affiliation":[{"name":"The University of Kansas Medical Center 1 Department of Pathology and Laboratory Medicine , , Kansas City, KS 66126 ,","place":["USA"]},{"name":"Institute of Reproductive and Developmental Sciences, The University of Kansas Medical Center 2 , Kansas City, KS 66126 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2025,4,28]]},"reference":[{"key":"2025042805003271100_DMM052222C1","doi-asserted-by":"publisher","first-page":"1511","DOI":"10.1007\/s00429-014-0741-9","article-title":"A novel form of ciliopathy underlies hyperphagia and obesity in Ankrd26 knockout mice","volume":"220","author":"Acs","year":"2015","journal-title":"Brain Struct. 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Cancer Prev."},{"key":"2025042805003271100_DMM052222C10","doi-asserted-by":"publisher","first-page":"7875","DOI":"10.1074\/jbc.M807827200","article-title":"Identification and functional analysis of a novel cyclin e\/cdk2 substrate ankrd17","volume":"284","author":"Deng","year":"2009","journal-title":"J. Biol. Chem."},{"key":"2025042805003271100_DMM052222C12","doi-asserted-by":"publisher","first-page":"1865","DOI":"10.3892\/ijmm.2018.3783","article-title":"Screening of circular RNAs and validation of circANKRD36 associated with inflammation in patients with type 2 diabetes mellitus","volume":"42","author":"Fang","year":"2018","journal-title":"Int. J. Mol. Med."},{"key":"2025042805003271100_DMM052222C13","doi-asserted-by":"publisher","first-page":"869-869","DOI":"10.1093\/Ajcp\/Aqaa163","article-title":"Robbins and cotran pathologic basis of disease","volume":"154","author":"Hoda","year":"2020","journal-title":"Am. J. Clin. 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