Lack of the human choline transporter-like protein SLC44A2 causes hearing impairment and a rare red blood phenotype.


Journal

EMBO molecular medicine
ISSN: 1757-4684
Titre abrégé: EMBO Mol Med
Pays: England
ID NLM: 101487380

Informations de publication

Date de publication:
08 03 2023
Historique:
revised: 06 01 2023
received: 17 05 2022
accepted: 10 01 2023
pubmed: 26 1 2023
medline: 11 3 2023
entrez: 25 1 2023
Statut: ppublish

Résumé

Blood phenotypes are defined by the presence or absence of specific blood group antigens at the red blood cell (RBC) surface, due to genetic polymorphisms among individuals. The recent development of genomic and proteomic approaches enabled the characterization of several enigmatic antigens. The choline transporter-like protein CTL2 encoded by the SLC44A2 gene plays an important role in platelet aggregation and neutrophil activation. By investigating alloantibodies to a high-prevalence antigen of unknown specificity, found in patients with a rare blood type, we showed that SLC44A2 is also expressed in RBCs and carries a new blood group system. Furthermore, we identified three siblings homozygous for a large deletion in SLC44A2, resulting in complete SLC44A2 deficiency. Interestingly, the first-ever reported SLC44A2-deficient individuals suffer from progressive hearing impairment, recurrent arterial aneurysms, and epilepsy. Furthermore, SLC44A2

Identifiants

pubmed: 36695047
doi: 10.15252/emmm.202216320
pmc: PMC9994479
doi:

Substances chimiques

choline transporter 0
Membrane Transport Proteins 0
SLC44A2 protein, human 0
Membrane Glycoproteins 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

e16320

Informations de copyright

© 2023 The Authors. Published under the terms of the CC BY 4.0 license.

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Auteurs

Bérengère Koehl (B)

Université Paris Cité and Université des Antilles, INSERM, BIGR, Paris, France.
Department of Child Hematology, Reference Center for Sickle-Cell Disease Robert Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.

Cédric Vrignaud (C)

Université Paris Cité and Université des Antilles, INSERM, BIGR, Paris, France.

Mahmoud Mikdar (M)

Université Paris Cité and Université des Antilles, INSERM, BIGR, Paris, France.

Thankam S Nair (TS)

Kresge Hearing Research Institute, Department of Otolaryngology/Head and Neck Surgery, University of Michigan, Ann Arbor, MI, USA.

Lucy Yang (L)

Kresge Hearing Research Institute, Department of Otolaryngology/Head and Neck Surgery, University of Michigan, Ann Arbor, MI, USA.

Seyve Landry (S)

Hemostasis Laboratory, Grenoble Alpes University Hospital Grenoble, Grenoble, France.

Guy Laiguillon (G)

Établissement Français de Sang (EFS) Ile-de-France, Centre National de Référence pour les Groupes Sanguins, Paris, France.

Claudine Giroux-Lathuile (C)

Etablissement Français du Sang Auvergne Rhône Alpes, Immunohematology Laboratory, Grenoble, France.

Sophie Anselme-Martin (S)

Etablissement Français du Sang Auvergne Rhône Alpes, Immunohematology Laboratory, Grenoble, France.

Hanane El Kenz (H)

Department of Transfusion, Blood Bank, CHU-Brugmann and Hôpital Universitaire des Enfants Reine Fabiola, Université Libre de Bruxelles, Brussels, Belgium.

Olivier Hermine (O)

Université de Paris, Imagine Institute, INSERM UMR 1163, Paris, France.

Narla Mohandas (N)

Red Cell Physiology Laboratory, New York Blood Center, New York, NY, USA.

Jean Pierre Cartron (JP)

Université Paris Cité and Université des Antilles, INSERM, BIGR, Paris, France.

Yves Colin (Y)

Université Paris Cité and Université des Antilles, INSERM, BIGR, Paris, France.

Olivier Detante (O)

Stroke Unit, Neurology Department, Grenoble Hospital, Grenoble Institute of Neurosciences, Inserm U1216, University of Grenoble Alpes, Grenoble, France.

Raphaël Marlu (R)

Hemostasis Laboratory, Grenoble Alpes University Hospital Grenoble, Grenoble, France.
University Grenoble Alpes, CNRS UMR5525, TIMC, Grenoble, France.

Caroline Le Van Kim (C)

Université Paris Cité and Université des Antilles, INSERM, BIGR, Paris, France.

Thomas E Carey (TE)

Kresge Hearing Research Institute, Department of Otolaryngology/Head and Neck Surgery, University of Michigan, Ann Arbor, MI, USA.

Slim Azouzi (S)

Université Paris Cité and Université des Antilles, INSERM, BIGR, Paris, France.
Établissement Français de Sang (EFS) Ile-de-France, Centre National de Référence pour les Groupes Sanguins, Paris, France.

Thierry Peyrard (T)

Université Paris Cité and Université des Antilles, INSERM, BIGR, Paris, France.
Établissement Français de Sang (EFS) Ile-de-France, Centre National de Référence pour les Groupes Sanguins, Paris, France.

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