| Literature DB >> 28748642 |
Paul Kruszka1, Antonio R Porras2, Yonit A Addissie1, Angélica Moresco3, Sofia Medrano3, Gary T K Mok4, Gordon K C Leung4, Cedrik Tekendo-Ngongang5, Annette Uwineza6, Meow-Keong Thong7, Premala Muthukumarasamy7, Engela Honey8, Ekanem N Ekure9, Ogochukwu J Sokunbi9, Nnenna Kalu9, Kelly L Jones10, Julie D Kaplan10, Omar A Abdul-Rahman10, Lisa M Vincent11, Amber Love11, Khadija Belhassan1,12, Karim Ouldim12, Ihssane El Bouchikhi12,13, Anju Shukla14, Katta M Girisha14, Siddaramappa J Patil15, Nirmala D Sirisena16, Vajira H W Dissanayake16, C Sampath Paththinige16, Rupesh Mishra16, Eva Klein-Zighelboim17, Bertha E Gallardo Jugo17, Miguel Chávez Pastor17, Hugo H Abarca-Barriga17, Steven A Skinner18, Eloise J Prijoles18, Eben Badoe19, Ashleigh D Gill1, Vorasuk Shotelersuk20, Patroula Smpokou21, Monisha S Kisling21, Carlos R Ferreira21, Leon Mutesa6, Andre Megarbane22, Antonie D Kline23, Amy Kimball23, Emmy Okello24, Peter Lwabi24, Twalib Aliku24, Emmanuel Tenywa24,25, Nonglak Boonchooduang26, Pranoot Tanpaiboon21, Antonio Richieri-Costa27, Ambroise Wonkam5, Brian H Y Chung4, Roger E Stevenson18, Marshall Summar21, Kausik Mandal28, Shubha R Phadke28, María G Obregon3, Marius G Linguraru2, Maximilian Muenke1.
Abstract
Noonan syndrome (NS) is a common genetic syndrome associated with gain of function variants in genes in the Ras/MAPK pathway. The phenotype of NS has been well characterized in populations of European descent with less attention given to other groups. In this study, individuals from diverse populations with NS were evaluated clinically and by facial analysis technology. Clinical data and images from 125 individuals with NS were obtained from 20 countries with an average age of 8 years and female composition of 46%. Individuals were grouped into categories of African descent (African), Asian, Latin American, and additional/other. Across these different population groups, NS was phenotypically similar with only 2 of 21 clinical elements showing a statistically significant difference. The most common clinical characteristics found in all population groups included widely spaced eyes and low-set ears in 80% or greater of participants, short stature in more than 70%, and pulmonary stenosis in roughly half of study individuals. Using facial analysis technology, we compared 161 Caucasian, African, Asian, and Latin American individuals with NS with 161 gender and age matched controls and found that sensitivity was equal to or greater than 94% for all groups, and specificity was equal to or greater than 90%. In summary, we present consistent clinical findings from global populations with NS and additionally demonstrate how facial analysis technology can support clinicians in making accurate NS diagnoses. This work will assist in earlier detection and in increasing recognition of NS throughout the world.Entities:
Keywords: Africa; Asia; Latin America; Middle East; Noonan syndrome; diverse populations; facial analysis technology
Mesh:
Substances:
Year: 2017 PMID: 28748642 PMCID: PMC5710841 DOI: 10.1002/ajmg.a.38362
Source DB: PubMed Journal: Am J Med Genet A ISSN: 1552-4825 Impact factor: 2.802