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Noonan syndrome in diverse populations.
Kruszka, Paul; Porras, Antonio R; Addissie, Yonit A; Moresco, Angélica; Medrano, Sofia; Mok, Gary T K; Leung, Gordon K C; Tekendo-Ngongang, Cedrik; Uwineza, Annette; Thong, Meow-Keong; Muthukumarasamy, Premala; Honey, Engela; Ekure, Ekanem N; Sokunbi, Ogochukwu J; Kalu, Nnenna; Jones, Kelly L; Kaplan, Julie D; Abdul-Rahman, Omar A; Vincent, Lisa M; Love, Amber; Belhassan, Khadija; Ouldim, Karim; El Bouchikhi, Ihssane; Shukla, Anju; Girisha, Katta M; Patil, Siddaramappa J; Sirisena, Nirmala D; Dissanayake, Vajira H W; Paththinige, C Sampath; Mishra, Rupesh; Klein-Zighelboim, Eva; Gallardo Jugo, Bertha E; Chávez Pastor, Miguel; Abarca-Barriga, Hugo H; Skinner, Steven A; Prijoles, Eloise J; Badoe, Eben; Gill, Ashleigh D; Shotelersuk, Vorasuk; Smpokou, Patroula; Kisling, Monisha S; Ferreira, Carlos R; Mutesa, Leon; Megarbane, Andre; Kline, Antonie D; Kimball, Amy; Okello, Emmy; Lwabi, Peter; Aliku, Twalib; Tenywa, Emmanuel.
Afiliação
  • Kruszka P; Medical Genetics Branch, National Human Genome Research Institute, The National Institutes of Health, Bethesda, Maryland.
  • Porras AR; Children's National Health System, Sheikh Zayed Institute for Pediatric Surgical Innovation, Washington, District of Columbia.
  • Addissie YA; Medical Genetics Branch, National Human Genome Research Institute, The National Institutes of Health, Bethesda, Maryland.
  • Moresco A; Servicio de Genética, Hospital de Pediatría Garrahan, Buenos Aires, Argentina.
  • Medrano S; Servicio de Genética, Hospital de Pediatría Garrahan, Buenos Aires, Argentina.
  • Mok GTK; LKS Faculty of Medicine, Department of Paediatrics and Adolescent Medicine, The University of Hong Kong, Hong Kong Special Administrative Region, Hong Kong, China.
  • Leung GKC; LKS Faculty of Medicine, Department of Paediatrics and Adolescent Medicine, The University of Hong Kong, Hong Kong Special Administrative Region, Hong Kong, China.
  • Tekendo-Ngongang C; Division of Human Genetics, University of Cape Town, Cape Town, South Africa.
  • Uwineza A; Center of Human Genetics, School of Medicine and Pharmacy, College of Medicine and Pharmacy, University of Rwanda, Kigali, Rwanda.
  • Thong MK; Faculty of Medicine,Department of Paediatrics, University of Malaya, Kuala Lumpur, Malaysia.
  • Muthukumarasamy P; Faculty of Medicine,Department of Paediatrics, University of Malaya, Kuala Lumpur, Malaysia.
  • Honey E; Department of Genetics, University of Pretoria, Pretoria, South Africa.
  • Ekure EN; Department of Paediatrics College of Medicine, University of Lagos, Lagos University Teaching Hospital, Lagos, Nigeria.
  • Sokunbi OJ; Department of Paediatrics College of Medicine, University of Lagos, Lagos University Teaching Hospital, Lagos, Nigeria.
  • Kalu N; Department of Paediatrics College of Medicine, University of Lagos, Lagos University Teaching Hospital, Lagos, Nigeria.
  • Jones KL; Division of Medical Genetics, Department of Pediatrics, University of Mississippi Medical Center, Jackson, Mississippi.
  • Kaplan JD; Division of Medical Genetics, Department of Pediatrics, University of Mississippi Medical Center, Jackson, Mississippi.
  • Abdul-Rahman OA; Division of Medical Genetics, Department of Pediatrics, University of Mississippi Medical Center, Jackson, Mississippi.
  • Vincent LM; GeneDx, Gaithersburg, Maryland.
  • Love A; GeneDx, Gaithersburg, Maryland.
  • Belhassan K; Medical Genetics Branch, National Human Genome Research Institute, The National Institutes of Health, Bethesda, Maryland.
  • Ouldim K; Medical Genetics and Oncogenetics Unit, Hassan II University Hospital, Fez, Morocco.
  • El Bouchikhi I; Medical Genetics and Oncogenetics Unit, Hassan II University Hospital, Fez, Morocco.
  • Shukla A; Medical Genetics and Oncogenetics Unit, Hassan II University Hospital, Fez, Morocco.
  • Girisha KM; Faculty of Sciences and Techniques,Laboratory of Microbial Biotechnology, University of Sidi Mohammed Ben Abdellah, Fez, Morocco.
  • Patil SJ; Department of Medical Genetics, Kasturba Medical College, Manipal University, Manipal, India.
  • Sirisena ND; Department of Medical Genetics, Kasturba Medical College, Manipal University, Manipal, India.
  • Dissanayake VHW; Mazumdar Shaw Medical Center, Narayana Health City, Bangalore, India.
  • Paththinige CS; Faculty of Medicine, Human Genetics Unit, University of Colombo, Colombo, Sri Lanka.
  • Mishra R; Faculty of Medicine, Human Genetics Unit, University of Colombo, Colombo, Sri Lanka.
  • Klein-Zighelboim E; Faculty of Medicine, Human Genetics Unit, University of Colombo, Colombo, Sri Lanka.
  • Gallardo Jugo BE; Faculty of Medicine, Human Genetics Unit, University of Colombo, Colombo, Sri Lanka.
  • Chávez Pastor M; Instituto Nacional de Salud del Niño, Lima, Peru.
  • Abarca-Barriga HH; Instituto Nacional de Salud del Niño, Lima, Peru.
  • Skinner SA; Instituto Nacional de Salud del Niño, Lima, Peru.
  • Prijoles EJ; Instituto Nacional de Salud del Niño, Lima, Peru.
  • Badoe E; Greenwood Genetic Center, Greenwood, South Carolina.
  • Gill AD; Greenwood Genetic Center, Greenwood, South Carolina.
  • Shotelersuk V; School of Medicine and Dentistry,Department of Child Health, College of Health Sciences, Accra, Ghana.
  • Smpokou P; Medical Genetics Branch, National Human Genome Research Institute, The National Institutes of Health, Bethesda, Maryland.
  • Kisling MS; Faculty of Medicine,Center of Excellence for Medical Genetics, Department of Pediatrics, Chulalongkorn University, Bangkok, Thailand.
  • Ferreira CR; Division of Genetics and Metabolism, Children's National Health System, Washington, District of Columbia.
  • Mutesa L; Division of Genetics and Metabolism, Children's National Health System, Washington, District of Columbia.
  • Megarbane A; Division of Genetics and Metabolism, Children's National Health System, Washington, District of Columbia.
  • Kline AD; Center of Human Genetics, School of Medicine and Pharmacy, College of Medicine and Pharmacy, University of Rwanda, Kigali, Rwanda.
  • Kimball A; Institut Jérôme Lejeune, Paris, France.
  • Okello E; Harvey Institute for Human Genetics, Greater Baltimore Medical Center, Baltimore, Maryland.
  • Lwabi P; Harvey Institute for Human Genetics, Greater Baltimore Medical Center, Baltimore, Maryland.
  • Aliku T; Uganda Heart Institute, Kampala, Uganda.
  • Tenywa E; Uganda Heart Institute, Kampala, Uganda.
Am J Med Genet A ; 173(9): 2323-2334, 2017 Sep.
Article em En | MEDLINE | ID: mdl-28748642
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.
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Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Face / Genética Populacional / Síndrome de Noonan Limite: Child / Female / Humans / Male Idioma: En Revista: Am J Med Genet A Assunto da revista: GENETICA MEDICA Ano de publicação: 2017 Tipo de documento: Article

Texto completo: 1 Coleções: 01-internacional Base de dados: MEDLINE Assunto principal: Face / Genética Populacional / Síndrome de Noonan Limite: Child / Female / Humans / Male Idioma: En Revista: Am J Med Genet A Assunto da revista: GENETICA MEDICA Ano de publicação: 2017 Tipo de documento: Article