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Barth syndrome (BTHS), a life-threatening disease typically diagnosed with cardiomyopathy and neutropenia, is caused by mutations in the mitochondrial transacylase tafazzin. By high-resolution<jats:sup>31<\/jats:sup>P nuclear magnetic resonance (NMR) with cryoprobe technology, recently we found a dramatic loss of choline plasmalogen in the tafazzin-knockdown (TAZ-KD) mouse heart, besides observing characteristic cardiolipin (CL) alterations in BTHS. In inner mitochondrial membrane where tafazzin locates, CL and diacyl phosphatidylethanolamine are known to be essential via lipid\u2013protein interactions reflecting their cone shape for integrity of respiratory chain supercomplexes and cristae ultrastructure. Here, we investigate the TAZ-KD brain, liver, kidney, and lymphoblast from patients compared with controls. We identified common yet markedly cell type\u2013dependent losses of ethanolamine plasmalogen as the dominant plasmalogen class therein. Tafazzin function thus critically relates to homeostasis of plasmalogen, which in the ethanolamine class has conceivably analogous and more potent molecular functions in mitochondria than diacyl phosphatidylethanolamine. The present discussion of a loss of plasmalogen\u2013protein interaction applies to other diseases with mitochondrial plasmalogen loss and aberrant forms of this organelle, including Alzheimer's disease.<\/jats:p>","DOI":"10.26508\/lsa.201900348","type":"journal-article","created":{"date-parts":[[2019,8,21]],"date-time":"2019-08-21T19:55:11Z","timestamp":1566417311000},"page":"e201900348","update-policy":"https:\/\/doi.org\/10.26508\/lsa.crossmarkpolicy","source":"Crossref","is-referenced-by-count":46,"title":["Plasmalogen loss caused by remodeling deficiency in mitochondria"],"prefix":"10.26508","volume":"2","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-4192-7852","authenticated-orcid":true,"given":"Tomohiro","family":"Kimura","sequence":"first","affiliation":[{"name":"Department of Biochemistry and Biomedical Sciences, McMaster University, Hamilton, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Atsuko 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