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A second cohort of CHD3 patients expands the molecular mechanisms known to cause Snijders Blok-Campeau syndrome

  • Theodore G. Drivas
  • , Dong Li
  • , Divya Nair
  • , Joseph T. Alaimo
  • , Mariëlle Alders
  • , Janine Altmüller
  • , Tahsin Stefan Barakat
  • , E. Martina Bebin
  • , Nicole L. Bertsch
  • , Patrick R. Blackburn
  • , Alyssa Blesson
  • , Arjan M. Bouman
  • , Knut Brockmann
  • , Perrine Brunelle
  • , Margit Burmeister
  • , Gregory M. Cooper
  • , Jonas Denecke
  • , Anne Dieux-Coëslier
  • , Holly Dubbs
  • , Alejandro Ferrer
  • Danna Gal, Lauren E. Bartik, Lauren B. Gunderson, Linda Hasadsri, Mahim Jain, Catherine Karimov, Beth Keena, Eric W. Klee, Katja Kloth, Baiba Lace, Marina Macchiaiolo, Julien L. Marcadier, Jeff M. Milunsky, Melanie P. Napier, Xilma R. Ortiz-Gonzalez, Pavel N. Pichurin, Jason Pinner, Zoe Powis, Chitra Prasad, Francesca Clementina Radio, Kristen J. Rasmussen, Deborah L. Renaud, Eric T. Rush, Carol Saunders, Duygu Selcen, Ann R. Seman, Deepali N. Shinde, Erica D. Smith, Thomas Smol, Lot Snijders Blok, Joan M. Stoler, Sha Tang, Marco Tartaglia, Michelle L. Thompson, Jiddeke M. van de Kamp, Jingmin Wang, Dagmar Weise, Karin Weiss, Rixa Woitschach, Bernd Wollnik, Huifang Yan, Elaine H. Zackai, Giuseppe Zampino, Philippe Campeau, Elizabeth Bhoj
  • The Children's Hospital of Philadelphia
  • University of Missouri at Kansas City
  • Children's Mercy Hospitals and Clinics
  • University of Amsterdam
  • University of Cologne
  • Erasmus University Rotterdam
  • University of Alabama at Birmingham
  • Mayo Clinic Rochester, MN
  • Kennedy Krieger Institute
  • University of Göttingen
  • Université de Lille
  • University of Michigan, Ann Arbor
  • HudsonAlpha Institute for Biotechnology
  • University of Hamburg
  • Technion-Israel Institute of Technology
  • Bone and Osteogenesis Imperfecta Department
  • University of Southern California
  • Université Laval
  • IRCCS Ospedale pediatrico Bambino Gesù - Roma
  • Alberta Children's Hospital
  • Center for Human Genetics
  • Western University
  • University of Pennsylvania
  • Sydney Children's Hospital
  • Ambry Genetics
  • University of Kansas
  • Boston Children's Hospital
  • Radboud University Nijmegen
  • Max Planck Institute for Psycholinguistics
  • Vrije Universiteit Amsterdam
  • Peking University
  • Rambam Health Care Campus
  • Fondazione Policlinico Universitario “A. Gemelli,” Universita Cattolica del Sacro Cuore
  • University of Montreal

Producción científicarevisión exhaustiva

39 Citas (Scopus)

Resumen

There has been one previous report of a cohort of patients with variants in Chromodomain Helicase DNA-binding 3 (CHD3), now recognized as Snijders Blok-Campeau syndrome. However, with only three previously-reported patients with variants outside the ATPase/helicase domain, it was unclear if variants outside of this domain caused a clinically similar phenotype. We have analyzed 24 new patients with CHD3 variants, including nine outside the ATPase/helicase domain. All patients were detected with unbiased molecular genetic methods. There is not a significant difference in the clinical or facial features of patients with variants in or outside this domain. These additional patients further expand the clinical and molecular data associated with CHD3 variants. Importantly we conclude that there is not a significant difference in the phenotypic features of patients with various molecular disruptions, including whole gene deletions and duplications, and missense variants outside the ATPase/helicase domain. This data will aid both clinical geneticists and molecular geneticists in the diagnosis of this emerging syndrome.

Idioma originalEnglish
Páginas (desde-hasta)1422-1431
Número de páginas10
PublicaciónEuropean Journal of Human Genetics
Volumen28
N.º10
DOI
EstadoPublished - 1 oct 2020
Publicado de forma externa

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