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American journal of medical genetics. Part A · 2017

22q11.2 deletion syndrome in diverse populations.

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Kruszka Paul, Addissie Yonit A, McGinn Daniel E, Porras Antonio R, Biggs Elijah, Share Matthew, Crowley T Blaine, Chung Brian H Y, Mok Gary T K, Mak Christopher C Y, Muthukumarasamy Premala, Thong Meow-Keong, Sirisena Nirmala D, Dissanayake Vajira H W, Paththinige C Sampath, Prabodha L B Lahiru, Mishra Rupesh, Shotelersuk Vorasuk, Ekure Ekanem Nsikak, Sokunbi Ogochukwu Jidechukwu, Kalu Nnenna, Ferreira Carlos R, Duncan Jordann-Mishael, Patil Siddaramappa Jagdish, Jones Kelly L, Kaplan Julie D, Abdul-Rahman Omar A, Uwineza Annette, Mutesa Leon, Moresco Angélica, Obregon María Gabriela, Richieri-Costa Antonio, Gil-da-Silva-Lopes Vera L, Adeyemo Adebowale A, Summar Marshall, Zackai Elaine H, McDonald-McGinn Donna M, Linguraru Marius George, Muenke Maximilian

Аннотация

22q11.2 deletion syndrome (22q11.2 DS) is the most common microdeletion syndrome and is underdiagnosed in diverse populations. This syndrome has a variable phenotype and affects multiple systems, making early recognition imperative. In this study, individuals from diverse populations with 22q11.2 DS were evaluated clinically and by facial analysis technology. Clinical information from 106 individuals and images from 101 were collected from individuals with 22q11.2 DS from 11 countries; average age was 11.7 and 47% were male. Individuals were grouped into categories of African descent (African), Asian, and Latin American. We found that the phenotype of 22q11.2 DS varied across population groups. Only two findings, congenital heart disease and learning problems, were found in greater than 50% of participants. When comparing the clinical features of 22q11.2 DS in each population, the proportion of individuals within each clinical category was statistically different except for learning problems and ear anomalies (P < 0.05). However, when Africans were removed from analysis, six additional clinical features were found to be independent of ethnicity (P ≥ 0.05). Using facial analysis technology, we compared 156 Caucasians, Africans, Asians, and Latin American individuals with 22q11.2 DS with 156 age and gender matched controls and found that sensitivity and specificity were greater than 96% for all populations. In summary, we present the varied findings from global populations with 22q11.2 DS and demonstrate how facial analysis technology can assist clinicians in making accurate 22q11.2 DS diagnoses. This work will assist in earlier detection and in increasing recognition of 22q11.2 DS throughout the world.

Предметные рубрики MeSH

ИП Урсолов Владлен Сергеевич

ИНН 500118482352 · ОГРНИП 314500117700049

Реквизиты и контакты