White-Sutton syndrome is a rare developmental disorder characterized by global developmental delay, intellectual disabilities (ID), and neurobehavioral abnormalities secondary to pathogenic POGZ variants. The purpose of our study was to describe the neurocognitive phenotype of an unbiased national cohort of patients with identified POGZ pathogenic variants.
This study is based on a French collaboration through the AnDDI-Rares network, and includes 19 patients from 18 families with POGZ pathogenic variants. All clinical data and neuropsychological tests were collected from medical files.
Among the 19 patients, 14 patients exhibited ID (6 mild, 5 moderate and 3 severe). The 5 remaining patients had learning disabilities and shared a similar neurocognitive profile, including language difficulties, dysexecutive syndrome, attention disorders, slowness and social difficulties. One patient evaluated for autism was found to have moderate autism spectrum disorder.
This study reveals that the cognitive phenotype of patients with POGZ pathogenic variants can range from learning disabilities to severe ID. It highlights that pathogenic variations in the same genes can be reported in a large spectrum of neurocognitive profiles, and that children with learning disabilities could benefit from next generation sequencing techniques.
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Aurore Garde
Jenny Cornaton
Arthur Sorlin
Sébastien Moutton
Claire Nicolas
Christine Juif
David Geneviève
Laurence Perrin
Philippe Khau-Van-Kien
Thomas Smol
Catherine Vincent-Delorme
Bertrand Isidor
Benjamin Cogne
Alexandra Afenjar
Boris Keren
Christine Coubes
Fabienne Prieur
Annick Toutain
Yann Trousselet
Solène Bourgouin
Coralie Gonin-Olympiade
Kim Giraudat
Amélie Piton
Bénédicte Gérard
Sylvie Odent
Fanny Tessier
Lola Lemasson
Solveig Heide
Anne-Claire Gelineau
Catherine Sarret
Anne Miret
Elise Schaefer
Juliette Piard
Rémi Mathevet
Marion Boucon
Ange-Line Bruel
Frederic Tran Mau-Them
Martin Chevarin
Antonio Vitobello
Christophe Philippe
Christel Thauvin-Robinet
Laurence Faivre
References
PubMed