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A Solve-RD ClinVar-based reanalysis of 1522 index cases from ERN-ITHACA reveals common pitfalls and misinterpretations in exome sequencing

Authors :
Denommé-Pichon, Anne-Sophie
Matalonga, Leslie
de Boer, Elke
Jackson, Adam
Benetti, Elisa
Banka, Siddharth
Bruel, Ange-Line
Ciolfi, Andrea
Clayton-Smith, Jill
Dallapiccola, Bruno
Duffourd, Yannis
Ellwanger, Kornelia
Fallerini, Chiara
Gilissen, Christian
Graessner, Holm
Haack, Tobias B.
Havlovicova, Marketa
Hoischen, Alexander
Jean-Marçais, Nolwenn
Kleefstra, Tjitske
López-Martín, Estrella
Macek, Milan
Mencarelli, Maria Antonietta
Moutton, Sébastien
Pfundt, Rolph
Pizzi, Simone
Posada, Manuel
Radio, Francesca Clementina
Renieri, Alessandra
Rooryck, Caroline
Ryba, Lukas
Safraou, Hana
Schwarz, Martin
Tartaglia, Marco
Thauvin-Robinet, Christel
Thevenon, Julien
Mau-Them, Frédéric Tran
Trimouille, Aurélien
Votypka, Pavel
de Vries, Bert B.A.
Willemsen, Marjolein H.
Zurek, Birte
Verloes, Alain
Philippe, Christophe
Abbott, Kristin M.
Banka, Siddharth
de Boer, Elke
Ciolfi, Andrea
Clayton-Smith, Jill
Dallapiccola, Bruno
Denommé-Pichon, Anne-Sophie
Faivre, Laurence
Gilissen, Christian
Haack, Tobias B.
Havlovicova, Marketa
Hoischen, Alexander
Jackson, Adam
Kerstjens, Mieke
Kleefstra, Tjitske
Martín, Estrella López
Macek, Milan
Matalonga, Leslie
Maystadt, Isabelle
Morleo, Manuela
Nigro, Vicenzo
Pinelli, Michele
Pizzi, Simone
Posada, Manuel
Radio, Francesca C.
Renieri, Alessandra
Riess, Olaf
Rooryck, Caroline
Ryba, Lukas
Agathe, Jean-Madeleine de Sainte
Santen, Gijs W.E.
Schwarz, Martin
Tartaglia, Marco
Thauvin, Christel
Torella, Annalaura
Trimouille, Aurélien
Verloes, Alain
Vissers, Lisenka
Vitobello, Antonio
Votypka, Pavel
Zguro, Kristina
Boer, Elke de
Cohen, Enzo
Danis, Daniel
Denommé-Pichon, Anne-Sophie
Gao, Fei
Gilissen, Christian
Horvath, Rita
Johari, Mridul
Johanson, Lennart
Li, Shuang
Matalonga, Leslie
Morsy, Heba
Nelson, Isabelle
Paramonov, Ida
te Paske, Iris B.A.W.
Robinson, Peter
Savarese, Marco
Steyaert, Wouter
Töpf, Ana
Trimouille, Aurélien
van der Velde, Joeri K.
Vandrovcova, Jana
Vitobello, Antonio
Riess, Olaf
Haack, Tobias B.
Graessner, Holm
Zurek, Birte
Ellwanger, Kornelia
Ossowski, Stephan
Demidov, German
Sturm, Marc
Schulze-Hentrich, Julia M.
Schüle, Rebecca
Xu, Jishu
Kessler, Christoph
Wayand, Melanie
Synofzik, Matthis
Wilke, Carlo
Traschütz, Andreas
Schöls, Ludger
Hengel, Holger
Lerche, Holger
Kegele, Josua
Heutink, Peter
Brunner, Han
Scheffer, Hans
Hoogerbrugge, Nicoline
Hoischen, Alexander
‘t Hoen, Peter A.C.
Vissers, Lisenka E.L.M.
Gilissen, Christian
Steyaert, Wouter
Sablauskas, Karolis
de Voer, Richarda M.
Kamsteeg, Erik-Jan
van de Warrenburg, Bart
van Os, Nienke
Paske, Iris te
Janssen, Erik
de Boer, Elke
Steehouwer, Marloes
Yaldiz, Burcu
Kleefstra, Tjitske
Brookes, Anthony J.
Veal, Colin
Gibson, Spencer
Maddi, Vatsalya
Mehtarizadeh, Mehdi
Riaz, Umar
Warren, Greg
Dizjikan, Farid Yavari
Shorter, Thomas
Töpf, Ana
Straub, Volker
Bettolo, Chiara Marini
Manera, Jordi Diaz
Hambleton, Sophie
Engelhardt, Karin
Clayton-Smith, Jill
Banka, Siddharth
Alexander, Elizabeth
Jackson, Adam
Faivre, Laurence
Thauvin, Christel
Vitobello, Antonio
Denommé-Pichon, Anne-Sophie
Duffourd, Yannis
Bruel, Ange-Line
Peyron, Christine
Pélissier, Aurore
Beltran, Sergi
Gut, Ivo Glynne
Laurie, Steven
Piscia, Davide
Matalonga, Leslie
Papakonstantinou, Anastasios
Bullich, Gemma
Corvo, Alberto
Fernandez-Callejo, Marcos
Hernández, Carles
Picó, Daniel
Paramonov, Ida
Lochmüller, Hanns
Gumus, Gulcin
Bros-Facer, Virginie
Rath, Ana
Hanauer, Marc
Lagorce, David
Hongnat, Oscar
Chahdil, Maroua
Lebreton, Emeline
Stevanin, Giovanni
Durr, Alexandra
Davoine, Claire-Sophie
Guillot-Noel, Léna
Heinzmann, Anna
Coarelli, Giulia
Bonne, Gisèle
Evangelista, Teresinha
Allamand, Valérie
Nelson, Isabelle
Ben Yaou, Rabah
Metay, Corinne
Eymard, Bruno
Cohen, Enzo
Atalaia, Antonio
Stojkovic, Tanya
Macek, Milan
Turnovec, Marek
Thomasová, Dana
Kremliková, Radka Pourová
Franková, Vera
Havlovicová, Markéta
Lišková, Petra
Doležalová, Pavla
Parkinson, Helen
Keane, Thomas
Freeberg, Mallory
Thomas, Coline
Spalding, Dylan
Robinson, Peter
Danis, Daniel
Robert, Glenn
Costa, Alessia
Patch, Christine
Hanna, Mike
Houlden, Henry
Reilly, Mary
Vandrovcova, Jana
Efthymiou, Stephanie
Morsy, Heba
Cali, Elisa
Magrinelli, Francesca
Sisodiya, Sanjay M.
Rohrer, Jonathan
Muntoni, Francesco
Zaharieva, Irina
Sarkozy, Anna
Timmerman, Vincent
Baets, Jonathan
de Vries, Geert
De Winter, Jonathan
Beijer, Danique
de Jonghe, Peter
Van de Vondel, Liedewei
De Ridder, Willem
Weckhuysen, Sarah
Nigro, Vincenzo
Mutarelli, Margherita
Morleo, Manuela
Pinelli, Michele
Varavallo, Alessandra
Banfi, Sandro
Torella, Annalaura
Musacchia, Francesco
Piluso, Giulio
Ferlini, Alessandra
Selvatici, Rita
Gualandi, Francesca
Bigoni, Stefania
Rossi, Rachele
Neri, Marcella
Aretz, Stefan
Spier, Isabel
Sommer, Anna Katharina
Peters, Sophia
Oliveira, Carla
Pelaez, Jose Garcia
Matos, Ana Rita
José, Celina São
Ferreira, Marta
Gullo, Irene
Fernandes, Susana
Garrido, Luzia
Ferreira, Pedro
Carneiro, Fátima
Swertz, Morris A.
Johansson, Lennart
van der Velde, Joeri K.
van der Vries, Gerben
Neerincx, Pieter B.
Ruvolo, David
Abbott, Kristin M.
Kerstjens Frederikse, Wilhemina S.
Zonneveld-Huijssoon, Eveline
Roelofs-Prins, Dieuwke
van Gijn, Marielle
Köhler, Sebastian
Metcalfe, Alison
Verloes, Alain
Drunat, Séverine
Heron, Delphine
Mignot, Cyril
Keren, Boris
Agathe, Jean-Madeleine de Sainte
Rooryck, Caroline
Lacombe, Didier
Trimouille, Aurelien
Capella, Gabriel
Valle, Laura
Holinski-Feder, Elke
Laner, Andreas
Steinke-Lange, Verena
Cilio, Maria-Roberta
Carpancea, Evelina
Depondt, Chantal
Lederer, Damien
Sznajer, Yves
Duerinckx, Sarah
Mary, Sandrine
Macaya, Alfons
Cazurro-Gutiérrez, Ana
Pérez-Dueñas, Belén
Munell, Francina
Jarava, Clara Franco
Masó, Laura Batlle
Marcé-Grau, Anna
Colobran, Roger
Hackman, Peter
Johari, Mridul
Savarese, Marco
Udd, Bjarne
Hemelsoet, Dimitri
Dermaut, Bart
Schuermans, Nika
Poppe, Bruce
Verdin, Hannah
Osorio, Andrés Nascimento
Depienne, Christel
Roos, Andreas
Maystadt, Isabelle
Cordts, Isabell
Deschauer, Marcus
Striano, Pasquale
Zara, Federico
Riva, Antonella
Iacomino, Michele
Uva, Paolo
Scala, Marcello
Scudieri, Paolo
Başak, Ayşe Nazlı
Claeys, Kristl
Boztug, Kaan
Haimel, Matthias
W.E, Gijs
Ruivenkamp, Claudia A.L.
Natera de Benito, Daniel
Lochmüller, Hanns
Thompson, Rachel
Polavarapu, Kiran
Grimbacher, Bodo
Zaganas, Ioannis
Kokosali, Evgenia
Lambros, Mathioudakis
Evangeliou, Athanasios
Spilioti, Martha
Kapaki, Elisabeth
Bourbouli, Mara
Ciolfi, Andrea
Dallapiccola, Bruno
Pizzi, Simone
Radio, Francesca Clementina
Tartaglia, Marco
Balicza, Peter
Molnar, Maria Judit
De la Paz, Manuel Posada
Sánchez, Eva Bermejo
Martín, Estrella López
Delgado, Beatriz Martínez
Alonso García de la Rosa, F. Javier
Schröck, Evelin
Rump, Andreas
Mei, Davide
Vetro, Annalisa
Balestrini, Simona
Guerrini, Renzo
Horvath, Rita
Chinnery, Patrick F.
Ratnaike, Thiloka
Gao, Fei
Schon, Katherine
Maver, Ales
Peterlin, Borut
Münchau, Alexander
Lohmann, Katja
Herzog, Rebecca
Pauly, Martje
May, Patrick
Beeson, David
Cossins, Judith
Renieri, Alessandra
Furini, Simone
Fallerini, Chiara
Benetti, Elisa
Afenjar, Alexandra
Goldenberg, Alice
Masurel, Alice
Phan, Alice
Dieux-Coeslier, Anne
Fargeot, Anne
Guerrot, Anne-Marie
Toutain, Annick
Molin, Arnaud
Sorlin, Arthur
Putoux, Audrey
Jouret, Béatrice
Laudier, Béatrice
Demeer, Bénédicte
Doray, Bérénice
Bonniaud, Bertille
Isidor, Bertrand
Gilbert-Dussardier, Brigitte
Leheup, Bruno
Reversade, Bruno
Paul, Carle
Vincent-Delorme, Catherine
Neiva, Cecilia
Poirsier, Céline
Quélin, Chloé
Chiaverini, Christine
Coubes, Christine
Francannet, Christine
Colson, Cindy
Desplantes, Claire
Wells, Constance
Goizet, Cyril
Lederer, Damien
Sanlaville, Damien
Amram, Daniel
Lehalle, Daphné
Geneviève, David
Heron, Delphine
Lacombe, Didier
Gaillard, Dominique
Zivi, Einat
Sarrazin, Elisabeth
Steichen, Elisabeth
Schaefer, Élise
Lacaze, Elodie
Jacquemin, Emmanuel
Bongers, Ernie
Kilic, Esra
Colin, Estelle
Giuliano, Fabienne
Prieur, Fabienne
Laffargue, Fanny
Morice-Picard, Fanny
Petit, Florence
Cartault, François
Feillet, François
Baujat, Geneviève
Morin, Gilles
Diene, Gwenaëlle
Journel, Hubert
Maystadt, Isabelle
Perthus, Isabelle
Lespinasse, James
Alessandri, Jean-Luc
Amiel, Jeanne
Martinovic, Jelena
Delanne, Julian
Albuisson, Juliette
Lambert, Laëtitia
Perrin, Laurence
Ousager, Lilian Bomme
Van Maldergem, Lionel
Pinson, Lucile
Ruaud, Lyse
Samimi, Mahtab
Bournez, Marie
Bonnet-Dupeyron, Marie Noëlle
Vincent, Marie
Jacquemont, Marie-Line
Cordier-Alex, Marie-Pierre
Gérard-Blanluet, Marion
Willems, Marjolaine
Spodenkiewicz, Marta
Doco-Fenzy, Martine
Rossi, Massimiliano
Renaud, Mathilde
Fradin, Mélanie
Mathieu, Michèle
Holder-Espinasse, Muriel H.
Houcinat, Nada
Hanna, Nadine
Leperrier, Nathalie
Chassaing, Nicolas
Philip, Nicole
Boute, Odile
Van Kien, Philippe Khau
Parent, Philippe
Bitoun, Pierre
Sarda, Pierre
Vabres, Pierre
Jouk, Pierre-Simon
Touraine, Renaud
El Chehadeh, Salima
Whalen, Sandra
Marlin, Sandrine
Passemard, Sandrine
Grotto, Sarah
Bellanger, Séverine Audebert
Blesson, Sophie
Nambot, Sophie
Naudion, Sophie
Lyonnet, Stanislas
Odent, Sylvie
Attie-Bitach, Tania
Busa, Tiffany
Drouin-Garraud, Valérie
Layet, Valérie
Bizaoui, Varoona
Cusin, Véronica
Capri, Yline
Alembik, Yves
Vitobello, Antonio
Vissers, Lisenka E.L.M.
Faivre, Laurence
Source :
Genetics in Medicine; 20230101, Issue: Preprints
Publication Year :
2023

Abstract

Within the Solve-RD project (https://solve-rd.eu/), the European Reference Network for Intellectual disability, TeleHealth, Autism, and Congenital Anomalies aimed to investigate whether a reanalysis of exomes from unsolved cases based on ClinVar annotations could establish additional diagnoses. We present the results of the “ClinVar low-hanging fruit” reanalysis, reasons for the failure of previous analyses, and lessons learned.

Details

Language :
English
ISSN :
10983600 and 15300366
Issue :
Preprints
Database :
Supplemental Index
Journal :
Genetics in Medicine
Publication Type :
Periodical
Accession number :
ejs61725633
Full Text :
https://doi.org/10.1016/j.gim.2023.100018