Mechanism of KMT5B haploinsufficiency in neurodevelopment in humans and mice.


Journal

Science advances
ISSN: 2375-2548
Titre abrégé: Sci Adv
Pays: United States
ID NLM: 101653440

Informations de publication

Date de publication:
10 03 2023
Historique:
entrez: 10 3 2023
pubmed: 11 3 2023
medline: 15 3 2023
Statut: ppublish

Résumé

Pathogenic variants in

Identifiants

pubmed: 36897941
doi: 10.1126/sciadv.ade1463
pmc: PMC10005179
doi:

Substances chimiques

Methyltransferases EC 2.1.1.-

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

eade1463

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Auteurs

Sarah E Sheppard (SE)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Unit on Vascular Malformations, Division of Intramural Research, Eunice Kennedy Shriver National Institute of Child Health and Human Development, Bethesda, MD, USA.

Laura Bryant (L)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Rochelle N Wickramasekara (RN)

Stessman Laboratory, Department of Pharmacology and Neuroscience, Creighton University Medical School, Omaha, NE, USA.
Molecular Diagnostic Laboratory, Boys Town National Research Hospital, Omaha, NE, USA.

Courtney Vaccaro (C)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Brynn Robertson (B)

Stessman Laboratory, Department of Pharmacology and Neuroscience, Creighton University Medical School, Omaha, NE, USA.

Jodi Hallgren (J)

Stessman Laboratory, Department of Pharmacology and Neuroscience, Creighton University Medical School, Omaha, NE, USA.

Jason Hulen (J)

Stessman Laboratory, Department of Pharmacology and Neuroscience, Creighton University Medical School, Omaha, NE, USA.

Cynthia J Watson (CJ)

Stessman Laboratory, Department of Pharmacology and Neuroscience, Creighton University Medical School, Omaha, NE, USA.

Victor Faundes (V)

Division of Evolution and Genomic Sciences, School of Biological Sciences, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK.
Laboratorio de Genética y Enfermedades Metabólicas, Instituto de Nutrición y Tecnología de los Alimentos (INTA), Universidad de Chile, Santiago, Chile.

Yannis Duffourd (Y)

Unité Fonctionnelle d'Innovation diagnostique des maladies rares, FHU-TRANSLAD, CHU Dijon Bourgogne, Dijon, France.

Pearl Lee (P)

Abramson Family Cancer Research Institute, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.

M Celeste Simon (MC)

Abramson Family Cancer Research Institute, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.

Xavier de la Cruz (X)

Vall d'Hebron Institute of Research (VHIR), Universitat Autònoma de Barcelona, Barcelona, Spain.
Institució Catalana de Recerca I Estudis Avançats (ICREA), Barcelona, Spain.

Natália Padilla (N)

Vall d'Hebron Institute of Research (VHIR), Universitat Autònoma de Barcelona, Barcelona, Spain.

Marco Flores-Mendez (M)

Raymond G. Perelman Center for Cellular and Molecular Therapeutics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Naiara Akizu (N)

Raymond G. Perelman Center for Cellular and Molecular Therapeutics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
Department of Pathology and Laboratory Medicine, University of Pennsylvania, Philadelphia, PA, USA.

Jacqueline Smiler (J)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
10x Genomics, Pleasanton, CA, USA.

Renata Pellegrino Da Silva (R)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Dong Li (D)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Michael March (M)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Abdias Diaz-Rosado (A)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Isabella Peixoto de Barcelos (I)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Zhao Xiang Choa (ZX)

Epithelial Epigenetics and Development Laboratory, A*STAR Skin Research Labs, Singapore, Singapore.
Department of Biochemistry, Yong Loo Lin School of Medicine, National University of Singapore, Singapore, Singapore.

Chin Yan Lim (CY)

Epithelial Epigenetics and Development Laboratory, A*STAR Skin Research Labs, Singapore, Singapore.
Department of Biochemistry, Yong Loo Lin School of Medicine, National University of Singapore, Singapore, Singapore.

Christèle Dubourg (C)

Laboratoire de Génétique Moléculaire et Génomique, Centre Hospitalier Universitaire de Rennes, Rennes 35033, France.

Hubert Journel (H)

Service de Génétique Médicale, Hopital Chubert, Vannes, Bretagne, France.

Florence Demurger (F)

Department of Clinical Genetics, Service de Génétique Clinique, Centre de Référence Maladies Rares Centre Labellisé Anomalies du Développement-Ouest, Centre Hospitalier Universitaire de Rennes, Rennes 35033, France.

Maureen Mulhern (M)

Department of Pathology, Columbia University Irving Medical Center, New York, NY, USA.
Department of Neurology, Columbia University Irving Medical Center, New York, NY, USA.

Cigdem Akman (C)

Department of Neurology, Columbia University Irving Medical Center, New York, NY, USA.

Natalie Lippa (N)

Institute for Genomic Medicine, Columbia University Irving Medical Center, New York, NY, USA.

Marisa Andrews (M)

Division of Genetics and Genomic Medicine, Department of Pediatrics, Washington University School of Medicine, St. Louis, MO, USA.

Dustin Baldridge (D)

Division of Genetics and Genomic Medicine, Department of Pediatrics, Washington University School of Medicine, St. Louis, MO, USA.

John Constantino (J)

Department of Psychiatry, Washington University School of Medicine, St. Louis, MO, USA.

Arie van Haeringen (A)

Department of Clinical Genetics, Leiden University Medical Center, Leiden, Netherlands.

Irina Snoeck-Streef (I)

Department of Child Neurology, University Medical Center Utrecht, Utrecht, Netherlands.

Penny Chow (P)

Department of Pediatrics, Division of Craniofacial Medicine, University of Washington, Seattle, WA, USA.

Anne Hing (A)

Department of Pediatrics, Division of Craniofacial Medicine, University of Washington, Seattle, WA, USA.

John M Graham (JM)

Medical Genetics, Department of Pediatrics, Cedars-Sinai Medical Center, UCLA School of Medicine, Los Angeles, CA, USA.

Margaret Au (M)

Medical Genetics, Department of Pediatrics, Cedars-Sinai Medical Center, UCLA School of Medicine, Los Angeles, CA, USA.

Laurence Faivre (L)

UFR Des Sciences de Santé, INSERM-Université de Bourgogne UMR1231 GAD "Génétique des Anomalies du Développement," FHU-TRANSLAD, Dijon, France.
Centre de Référence Anomalies du Développement et Syndromes Malformatifs, CHU Dijon, Bourgogne, France.

Wei Shen (W)

University of Utah, Salt Lake City, UT, USA.
Mayo Clinic, Rochester, MN, USA.

Rong Mao (R)

University of Utah, Salt Lake City, UT, USA.

Janice Palumbos (J)

University of Utah, Salt Lake City, UT, USA.

David Viskochil (D)

University of Utah, Salt Lake City, UT, USA.

William Gahl (W)

NIH Undiagnosed Diseases Program, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD, USA.

Cynthia Tifft (C)

NIH Undiagnosed Diseases Program, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD, USA.

Ellen Macnamara (E)

NIH Undiagnosed Diseases Program, National Human Genome Research Institute, National Institutes of Health, Bethesda, MD, USA.

Natalie Hauser (N)

Medical Genetics, Inova Fairfax Hospital, Falls Church, VA, USA.

Rebecca Miller (R)

Medical Genetics, Inova Fairfax Hospital, Falls Church, VA, USA.

Jessica Maffeo (J)

Medical Genetics, Inova Fairfax Hospital, Falls Church, VA, USA.

Alexandra Afenjar (A)

AP-HP, Sorbonne Université, Département de neuropediatrie, Hospital Armand Trousseau, Paris, France.

Diane Doummar (D)

AP-HP, Sorbonne Université, Département de neuropediatrie, Hospital Armand Trousseau, Paris, France.

Boris Keren (B)

Genetic Department, Pitié-Salpêtrière Hospital, AP-HP, Sorbonne Université, Paris, France.

Pamela Arn (P)

Department of Pediatrics, Nemours Children's Specialty Care, Jacksonville, FL, USA.

Sarah Macklin-Mantia (S)

Department of Clinical Genomics, Mayo Clinic Florida, Jacksonville, FL, USA.

Ilse Meerschaut (I)

Center for Medical Genetics, Ghent University Hospital, Ghent, Belgium.

Bert Callewaert (B)

Center for Medical Genetics, Ghent University Hospital, Ghent, Belgium.
Department of Biomolecular Medicine, Ghent University, Ghent, Belgium.

André Reis (A)

Institute of Human Genetics, Friedrich-Alexander-Universität Erlangen-Nürnberg, 91054 Erlangen, Germany.

Christiane Zweier (C)

Institute of Human Genetics, Friedrich-Alexander-Universität Erlangen-Nürnberg, 91054 Erlangen, Germany.
Department of Human Genetics, Inselspital, Bern University Hospital, University of Bern, 3010 Bern, Switzerland.

Carole Brewer (C)

Clinical Genetics Department, Royal Devon and Exeter Hospital (Heavitree), Exeter EX1 2ED, UK.

Anand Saggar (A)

Clinical Genetics Department, St George's Hospital, St George's Healthcare NHS Trust, London SW17 0QT, UK.

Marie F Smeland (MF)

Department of Medical Genetics, University Hospital of North Norway, Tromsø, Norway.
Department of Pediatric Rehabilitation, University Hospital of North Norway, Norway.

Ajith Kumar (A)

Northeast Thames Regional Genetics Service, Great Ormond Street Hospital, London WC1N 3JH, UK.

Frances Elmslie (F)

South West Thames Centre for Genomics, St George's University Hospitals NHS Foundation Trust, London SW17 0QT, UK.

Charu Deshpande (C)

Department of Medical Genetics, Guy's Hospital, London SE1 9RT, UK.

Mathilde Nizon (M)

CHU Nantes, Service de Génétique Médicale, 9 quai Moncousu, 44093 Nantes CEDEX 1, France.

Benjamin Cogne (B)

CHU Nantes, Service de Génétique Médicale, 9 quai Moncousu, 44093 Nantes CEDEX 1, France.
Nantes Université, CNRS, INSERM, L'institut du thorax, F-44000 Nantes, France.

Yvette van Ierland (Y)

Department of Clinical Genetics, Erasmus University Medical Center, P.O. Box 2040, 3000 CA Rotterdam, Netherlands.

Martina Wilke (M)

Department of Clinical Genetics, Erasmus University Medical Center, P.O. Box 2040, 3000 CA Rotterdam, Netherlands.

Marjon van Slegtenhorst (M)

Department of Clinical Genetics, Erasmus University Medical Center, P.O. Box 2040, 3000 CA Rotterdam, Netherlands.

Suzanne Koudijs (S)

Department of Neurology, Erasmus University Medical Center-Sophia Children's Hospital, P.O. Box 2040, 3000 CA Rotterdam, Netherlands.

Jin Yun Chen (JY)

Neurology Department, Massachusetts General Hospital, Boston, MA, USA.

David Dredge (D)

University Children's Hospital Salzburg, Paracelsus Medical University (PMU), Salzburg, Austria.

Danielle Pier (D)

Neurology Department, Massachusetts General Hospital, Boston, MA, USA.

Saskia Wortmann (S)

University Children's Hospital Salzburg, Paracelsus Medical University (PMU), Salzburg, Austria.
Amalia Children's Hospital, RadboudUMC Nijmegen, Nijmegen, Netherlands.

Erik-Jan Kamsteeg (EJ)

University Children's Hospital Salzburg, Paracelsus Medical University (PMU), Salzburg, Austria.

Johannes Koch (J)

University Children's Hospital Salzburg, Paracelsus Medical University (PMU), Salzburg, Austria.

Devon Haynes (D)

Division of Genetics, Arnold Palmer Hospital for Children-Orlando Health, Orlando, FL, USA.

Lynda Pollack (L)

Division of Genetics, Arnold Palmer Hospital for Children-Orlando Health, Orlando, FL, USA.

Hannah Titheradge (H)

West Midlands Regional Genetics Service and Birmingham Health Partners, Birmingham Women's and Children's NHS Trust, Birmingham B15 2TG, UK.

Kara Ranguin (K)

Department of Genetics, Reference Centre for Rare Diseases and Developmental Anomalies, Caen Hospital, Caen, France.

Anne-Sophie Denommé-Pichon (AS)

Unité Fonctionnelle d'Innovation diagnostique des maladies rares, FHU-TRANSLAD, CHU Dijon Bourgogne, Dijon, France.
UFR Des Sciences de Santé, INSERM-Université de Bourgogne UMR1231 GAD "Génétique des Anomalies du Développement," FHU-TRANSLAD, Dijon, France.

Sacha Weber (S)

Department of Genetics, Reference Centre for Rare Diseases and Developmental Anomalies, Caen Hospital, Caen, France.

Rubén Pérez de la Fuente (R)

UDISGEN (Unidad de Dismorfología y Genética) 12 de Octubre University Hospital, Madrid, Spain.

Jaime Sánchez Del Pozo (J)

UDISGEN (Unidad de Dismorfología y Genética) 12 de Octubre University Hospital, Madrid, Spain.

Jose Miguel Lezana Rosales (JM)

UDISGEN (Unidad de Dismorfología y Genética) 12 de Octubre University Hospital, Madrid, Spain.

Pascal Joset (P)

University of Zurich, Institute of Medical Genetics, 8952 Schlieren-Zurich, Switzerland.

Katharina Steindl (K)

University of Zurich, Institute of Medical Genetics, 8952 Schlieren-Zurich, Switzerland.

Anita Rauch (A)

University of Zurich, Institute of Medical Genetics, 8952 Schlieren-Zurich, Switzerland.
University of Zurich, University Children's Hospital Zurich, 8032 Zurich, Switzerland.
University of Zurich, URPP Adaptive Brain Circuits in Development and Learning (AdaBD), Zurich, Switzerland.
University of Zurich Research Priority Program (URPP) AdaBD: Adaptive Brain Circuits in Development and Learning, Zurich 8006, Switzerland.
University of Zurich Research Priority Program (URPP) ITINERARE: Innovative Therapies in Rare Diseases, Zurich 8006, Switzerland.

Davide Mei (D)

Pediatric Neurology, Neurogenetics and Neurobiology Unit and Laboratories, Meyer Children's Hospital, Member of ERN Epicare, University of Florence, Florence, Italy.

Francesco Mari (F)

Pediatric Neurology, Neurogenetics and Neurobiology Unit and Laboratories, Meyer Children's Hospital, Member of ERN Epicare, University of Florence, Florence, Italy.

Renzo Guerrini (R)

Pediatric Neurology, Neurogenetics and Neurobiology Unit and Laboratories, Meyer Children's Hospital, Member of ERN Epicare, University of Florence, Florence, Italy.

James Lespinasse (J)

UF de Génétique Chromosomique, Centre Hospitalier de Chambéry, Hôtel-dieu, France.

Frédéric Tran Mau-Them (F)

Unité Fonctionnelle d'Innovation diagnostique des maladies rares, FHU-TRANSLAD, CHU Dijon Bourgogne, Dijon, France.
UFR Des Sciences de Santé, INSERM-Université de Bourgogne UMR1231 GAD "Génétique des Anomalies du Développement," FHU-TRANSLAD, Dijon, France.

Christophe Philippe (C)

Unité Fonctionnelle d'Innovation diagnostique des maladies rares, FHU-TRANSLAD, CHU Dijon Bourgogne, Dijon, France.
UFR Des Sciences de Santé, INSERM-Université de Bourgogne UMR1231 GAD "Génétique des Anomalies du Développement," FHU-TRANSLAD, Dijon, France.

Benjamin Dauriat (B)

Service de cytogénétique et génétique médicale, Centre Hospitalier Universitaire de Limoges, France.

Laure Raymond (L)

Service de génétique, Laboratoire Eurofins Biomnis, Lyon, France.

Sébastien Moutton (S)

Service de génétique, Laboratoire Eurofins Biomnis, Lyon, France.

Anna M Cueto-González (AM)

Hospital Vall d'Hebron, Barcelona, Spain.
Department of Clinical and Molecular Genetics, Vall d'Hebron Barcelona Hospital Campus, Passeig Vall d'Hebron 119-129, 08035 Barcelona, Spain.

Tiong Yang Tan (TY)

Victorian Clinical Genetics Services, Murdoch Children's Research Institute, Melbourne, VIC, Australia.
Department of Paediatrics, University of Melbourne, Melbourne, VIC, Australia.

Cyril Mignot (C)

AP-HP, Sorbonne Université, Département de Génétique, Paris, France.

Sarah Grotto (S)

AP-HP, Sorbonne Université, Département de Génétique, Paris, France.

Florence Renaldo (F)

AP-HP, Sorbonne Université, Département de neuropediatrie, Centre de référence neurogénétique, Hôpital Armand Trousseau, Paris, France.

Theodore G Drivas (TG)

Department of Genetics, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA.
Division of Translational Medicine and Human Genetics, Department of Medicine, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA.

Laura Hennessy (L)

Division of Translational Medicine and Human Genetics, Department of Medicine, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA.

Anna Raper (A)

Division of Translational Medicine and Human Genetics, Department of Medicine, Perelman School of Medicine at the University of Pennsylvania, Philadelphia, PA, USA.

Ilaria Parenti (I)

Institut für Humangenetik, Universitätsklinikum Essen, Universität Duisburg-Essen, Essen, Germany.

Frank J Kaiser (FJ)

Institut für Humangenetik, Universitätsklinikum Essen, Universität Duisburg-Essen, Essen, Germany.
Essener Zentrum für Seltene Erkrankungen (EZSE), Universitätsklinikum Essen, Essen, Germany.

Alma Kuechler (A)

Institut für Humangenetik, Universitätsklinikum Essen, Universität Duisburg-Essen, Essen, Germany.

Øyvind L Busk (ØL)

Department of Medical Genetics, Telemark Hospital Trust, 3710 Skien, Norway.

Lily Islam (L)

West Midlands Regional Genetics Service and Birmingham Health Partners, Birmingham Women's and Children's NHS Trust, Birmingham B15 2TG, UK.

Jacob A Siedlik (JA)

Department of Exercise Science and Pre-Health Professions, Creighton University, Omaha, NE, USA.

Lindsay B Henderson (LB)

GeneDx, Gaithersburg, MD, USA.

Jane Juusola (J)

GeneDx, Gaithersburg, MD, USA.

Richard Person (R)

GeneDx, Gaithersburg, MD, USA.

Rhonda E Schnur (RE)

GeneDx, Gaithersburg, MD, USA.
Department of Pediatrics, Division of Genetics Cooper Medical School of Rowan University Cooper University Health Care 3, Cooper Plaza, Camden, NJ, USA.

Antonio Vitobello (A)

Unité Fonctionnelle d'Innovation diagnostique des maladies rares, FHU-TRANSLAD, CHU Dijon Bourgogne, Dijon, France.
UFR Des Sciences de Santé, INSERM-Université de Bourgogne UMR1231 GAD "Génétique des Anomalies du Développement," FHU-TRANSLAD, Dijon, France.

Siddharth Banka (S)

Division of Evolution and Genomic Sciences, School of Biological Sciences, Faculty of Biology, Medicine and Health, University of Manchester, Manchester, UK.

Elizabeth J Bhoj (EJ)

Center for Applied Genomics, Children's Hospital of Philadelphia, Philadelphia, PA, USA.

Holly A F Stessman (HAF)

Stessman Laboratory, Department of Pharmacology and Neuroscience, Creighton University Medical School, Omaha, NE, USA.

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