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. 2020 Sep 15;11:652. doi: 10.3389/fendo.2020.00652

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Copyright © 2020 Kovilakath, Jamil and Cowart.

This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

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(A) Sphingolipid de novo synthesis. Overview of sphingolipid structure and metabolism. ssSPTa, small SPT subunit a; ssSPTb, small SPT subunit b; DHS, dihydrosphingosine; CerS, ceramide synthase; DHC, dihydroceramide; DES, DHC desaturase; SM, sphingomyelin; GSL, glycosphingolipid. (B) Sphingolipids involved in cardiogenesis. The receptors S1PR1-3, Spns2, OGR1, GPR12 and the sphingolipids S1P and SPC are highly expressed in stem cells that migrate to form the primitive heart tube. Normal expression of S1P via the S1PR1 receptor is needed for normal heart looping. Ceramide and S1P via the Cert and S1PR3 receptors, respectively, are required for chamber and septal formation. Cardia bifida is observed after failure of the myocardial cells to coalesce into one single primitive heart tube. Overexpression of Spns2 or S1PR1 or mutation of S1PR2 causes cardia bifida.