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Biallelic variation in the choline and ethanolamine transporter FLVCR1underlies a severe developmental disorder spectrum

Authors :
Calame, Daniel G.
Wong, Jovi Huixin
Panda, Puravi
Nguyen, Dat Tuan
Leong, Nancy C.P.
Sangermano, Riccardo
Patankar, Sohil G.
Abdel-Hamid, Mohamed S.
AlAbdi, Lama
Safwat, Sylvia
Flannery, Kyle P.
Dardas, Zain
Fatih, Jawid M.
Murali, Chaya
Kannan, Varun
Lotze, Timothy E.
Herman, Isabella
Ammouri, Farah
Rezich, Brianna
Efthymiou, Stephanie
Alavi, Shahryar
Murphy, David
Firoozfar, Zahra
Nasab, Mahya Ebrahimi
Bahreini, Amir
Ghasemi, Majid
Haridy, Nourelhoda A.
Goldouzi, Hamid Reza
Eghbal, Fatemeh
Karimiani, Ehsan Ghayoor
Begtrup, Amber
Elloumi, Houda
Srinivasan, Varunvenkat M.
Gowda, Vykuntaraju K.
Du, Haowei
Jhangiani, Shalini N.
Coban-Akdemir, Zeynep
Marafi, Dana
Rodan, Lance
Isikay, Sedat
Rosenfeld, Jill A.
Ramanathan, Subhadra
Staton, Michael
Oberg, Kerby C.
Clark, Robin D.
Wenman, Catharina
Loughlin, Sam
Saad, Ramy
Ashraf, Tazeen
Male, Alison
Tadros, Shereen
Boostani, Reza
Abdel-Salam, Ghada M.H.
Zaki, Maha
Mardi, Ali
Hashemi-Gorji, Farzad
Abdalla, Ebtesam
Manzini, M. Chiara
Pehlivan, Davut
Posey, Jennifer E.
Gibbs, Richard A.
Houlden, Henry
Alkuraya, Fowzan S.
Bujakowska, Kinga
Maroofian, Reza
Lupski, James R.
Nguyen, Long Nam
Source :
Genetics in Medicine; 20240101, Issue: Preprints
Publication Year :
2024

Abstract

FLVCR1encodes a solute carrier protein implicated in heme, choline, and ethanolamine transport. Although Flvcr1−/−mice exhibit skeletal malformations and defective erythropoiesis reminiscent of Diamond-Blackfan anemia (DBA), biallelic FLVCR1variants in humans have previously only been linked to childhood or adult-onset ataxia, sensory neuropathy, and retinitis pigmentosa.

Details

Language :
English
ISSN :
10983600 and 15300366
Issue :
Preprints
Database :
Supplemental Index
Journal :
Genetics in Medicine
Publication Type :
Periodical
Accession number :
ejs67436561
Full Text :
https://doi.org/10.1016/j.gim.2024.101273