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American journal of medical genetics. Part A · 2018

Williams-Beuren syndrome in diverse populations.

СтатьяМетаанализResearch Support, N.I.H., IntramuralПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод в очереди
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Kruszka Paul, Porras Antonio R, de Souza Deise Helena, Moresco Angélica, Huckstadt Victoria, Gill Ashleigh D, Boyle Alec P, Hu Tommy, Addissie Yonit A, Mok Gary T K, Tekendo-Ngongang Cedrik, Fieggen Karen, Prijoles Eloise J, Tanpaiboon Pranoot, Honey Engela, Luk Ho-Ming, Lo Ivan F M, Thong Meow-Keong, Muthukumarasamy Premala, Jones Kelly L, Belhassan Khadija, Ouldim Karim, El Bouchikhi Ihssane, Bouguenouch Laila, Shukla Anju, Girisha Katta M, Sirisena Nirmala D, Dissanayake Vajira H W, Paththinige C Sampath, Mishra Rupesh, Kisling Monisha S, Ferreira Carlos R, de Herreros María Beatriz, Lee Ni-Chung, Jamuar Saumya S, Lai Angeline, Tan Ee Shien, Ying Lim Jiin, Wen-Min Cham Breana, Gupta Neerja, Lotz-Esquivel Stephanie, Badilla-Porras Ramsés, Hussen Dalia Farouk, El Ruby Mona O, Ashaat Engy A, Patil Siddaramappa J, Dowsett Leah, Eaton Alison, Innes A Micheil, Shotelersuk Vorasuk, Badoe Ëben, Wonkam Ambroise, Obregon María Gabriela, Chung Brian H Y, Trubnykova Milana, La Serna Jorge, Gallardo Jugo Bertha Elena, Chávez Pastor Miguel, Abarca Barriga Hugo Hernán, Megarbane Andre, Kozel Beth A, van Haelst Mieke M, Stevenson Roger E, Summar Marshall, Adeyemo A Adebowale, Morris Colleen A, Moretti-Ferreira Danilo, Linguraru Marius George, Muenke Maximilian

Аннотация

Williams-Beuren syndrome (WBS) is a common microdeletion syndrome characterized by a 1.5Mb deletion in 7q11.23. The phenotype of WBS has been well described in populations of European descent with not as much attention given to other ethnicities. In this study, individuals with WBS from diverse populations were assessed clinically and by facial analysis technology. Clinical data and images from 137 individuals with WBS were found in 19 countries with an average age of 11 years and female gender of 45%. The most common clinical phenotype elements were periorbital fullness and intellectual disability which were present in greater than 90% of our cohort. Additionally, 75% or greater of all individuals with WBS had malar flattening, long philtrum, wide mouth, and small jaw. Using facial analysis technology, we compared 286 Asian, African, Caucasian, and Latin American individuals with WBS with 286 gender and age matched controls and found that the accuracy to discriminate between WBS and controls was 0.90 when the entire cohort was evaluated concurrently. The test accuracy of the facial recognition technology increased significantly when the cohort was analyzed by specific ethnic population (P-value < 0.001 for all comparisons), with accuracies for Caucasian, African, Asian, and Latin American groups of 0.92, 0.96, 0.92, and 0.93, respectively. In summary, we present consistent clinical findings from global populations with WBS and demonstrate how facial analysis technology can support clinicians in making accurate WBS diagnoses.

Предметные рубрики MeSH

ИП Урсолов Владлен Сергеевич

ИНН 500118482352 · ОГРНИП 314500117700049

Реквизиты и контакты