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Nearly all MYH9-RD patients present with macrothrombocytopenia, characterized by decreased platelet count and increased platelet size. In the present study, we collected blood samples from three MYH9-RD patients (R702S, D1424N, and R1464C) and unexpectedly found that the actin levels in the red blood cells (RBCs) from all three MYH9-RD patients are substantially lower than the healthy controls. We further revealed that the levels of two RBC membrane skeleton proteins, \u03b1-spectrin and tropomodulin, are also reduced in MYH9-RD RBCs. We showed that the membrane skeleton of MYH9-RD RBCs was more porous and that MYH9-RD RBCs produced more severe deformation under hyperosmotic pressure compared with healthy controls. We propose that defects in the membrane-skeleton network of RBCs may be an abnormal manifestation of MYH9-RD.<\/jats:p>","DOI":"10.1042\/bsr20260386","type":"journal-article","created":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T09:43:03Z","timestamp":1789638183000},"update-policy":"https:\/\/doi.org\/10.1042\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["The membrane skeleton density of red blood cells in\n                    <i>MYH9<\/i>\n                    -related disease patients is decreased"],"prefix":"10.1042","volume":"46","author":[{"given":"Shaopeng","family":"Sun","sequence":"first","affiliation":[{"name":"1State Key Laboratory of Animal Biodiversity Conservation and Integrated Pest Management, Institute of Zoology, Chinese Academy of Sciences, Beijing 100101, China"},{"name":"2University of Chinese Academy of Sciences, Beijing 100049, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jiabin","family":"Pan","sequence":"additional","affiliation":[{"name":"1State Key Laboratory of Animal Biodiversity Conservation and Integrated Pest Management, Institute of Zoology, Chinese Academy of Sciences, Beijing 100101, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ning","family":"Zhang","sequence":"additional","affiliation":[{"name":"1State Key Laboratory of Animal Biodiversity Conservation and Integrated Pest Management, Institute of Zoology, Chinese Academy of Sciences, Beijing 100101, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xianghong","family":"Jin","sequence":"additional","affiliation":[{"name":"3Department of Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tie-nan","family":"Zhu","sequence":"additional","affiliation":[{"name":"4Department of Hematology, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences & Peking Union Medical College, Beijing 100730, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhijie","family":"Kang","sequence":"additional","affiliation":[{"name":"5Department of Hematology, the Second Hospital of Dalian Medical University; Liaoning Medical Center for Hematopoietic Stem Cell Transplantation; Liaoning Key Laboratory of Hematopoietic Stem Cell Transplantation and Translational Medicine, Dalian, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jihong","family":"Hao","sequence":"additional","affiliation":[{"name":"6Department of Clinical Laboratory, the Second Hospital of Hebei Medical University, Shijiazhuang 050000, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8677-9833","authenticated-orcid":false,"given":"Xiang-dong","family":"Li","sequence":"additional","affiliation":[{"name":"1State Key Laboratory of Animal Biodiversity Conservation and Integrated Pest Management, Institute of Zoology, Chinese Academy of Sciences, Beijing 100101, China"},{"name":"2University of Chinese Academy of Sciences, Beijing 100049, China"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"288","published-online":{"date-parts":[[2026,9,29]]},"reference":[{"key":"2026092901415367600_B1","doi-asserted-by":"publisher","first-page":"189","DOI":"10.1055\/s-0029-1220327","article-title":"MYH9-related platelet disorders","volume":"35","author":"Althaus","year":"2009","journal-title":"Semin. 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