| Literature DB >> 34803161 |
Flavien Rouxel1, Kevin Yauy1, Guilaine Boursier2, Vincent Gatinois3, Mouna Barat-Houari2, Elodie Sanchez1, Didier Lacombe4, Stéphanie Arpin5, Fabienne Giuliano6, Damien Haye7,8, Marlène Rio9, Annick Toutain5, Klaus Dieterich10, Elise Brischoux-Boucher11, Sophie Julia12, Mathilde Nizon13, Alexandra Afenjar14, Boris Keren8, Aurelia Jacquette8, Sebastien Moutton15, Marie-Line Jacquemont16, Claire Duflos17, Yline Capri7, Jeanne Amiel9, Patricia Blanchet1, Stanislas Lyonnet9, Damien Sanlaville18, David Genevieve19.
Abstract
Kabuki syndrome (KS) is a rare genetic disorder caused by mutations in two major genes, KMT2D and KDM6A, that are responsible for Kabuki syndrome 1 (KS1, OMIM147920) and Kabuki syndrome 2 (KS2, OMIM300867), respectively. We lack a description of clinical signs to distinguish KS1 and KS2. We used facial morphology analysis to detect any facial morphological differences between the two KS types. We used a facial-recognition algorithm to explore any facial morphologic differences between the two types of KS. We compared several image series of KS1 and KS2 individuals, then compared images of those of Caucasian origin only (12 individuals for each gene) because this was the main ethnicity in this series. We also collected 32 images from the literature to amass a large series. We externally validated results obtained by the algorithm with evaluations by trained clinical geneticists using the same set of pictures. Use of the algorithm revealed a statistically significant difference between each group for our series of images, demonstrating a different facial morphotype between KS1 and KS2 individuals (mean area under the receiver operating characteristic curve = 0.85 [p = 0.027] between KS1 and KS2). The algorithm was better at discriminating between the two types of KS with images from our series than those from the literature (p = 0.0007). Clinical geneticists trained to distinguished KS1 and KS2 significantly recognised a unique facial morphotype, which validated algorithm findings (p = 1.6e-11). Our deep-neural-network-driven facial-recognition algorithm can reveal specific composite gestalt images for KS1 and KS2 individuals.Entities:
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Year: 2021 PMID: 34803161 PMCID: PMC9177756 DOI: 10.1038/s41431-021-00994-8
Source DB: PubMed Journal: Eur J Hum Genet ISSN: 1018-4813 Impact factor: 5.351