Literature DB >> 33510600

Clinical and Molecular Spectrum of Four Patients Diagnosed with Mowat-Wilson Syndrome.

Durdugul Ayyildiz Emecen1, Esra Isik1, Gulen E Utine2, Pelin O Simsek-Kiper2, Tahir Atik1, Ferda Ozkinay1.   

Abstract

Mowat-Wilson syndrome (MWS) is a rare autosomal dominant syndrome characterized by distinctive facial features, congenital heart defects, Hirschsprung disease, genitourinary anomalies, various structural brain anomalies, and intellectual disability. Pathogenic mutations that result in haploinsufficiency in the ZEB2 gene cause MWS. In this study, we aimed to evaluate the clinical features and molecular analysis results of 4 MWS patients. All patients were examined by an expert clinical geneticist. Dysmorphological abnormalities were recorded. Data including demographic, clinical, and laboratory findings were obtained from hospital records. ZEB2 gene analysis was performed using a Sanger sequencing method. All patients had typical facial features of MWS such as widely spaced eyes, broad eyebrows with a medial flare, low-hanging columella, prominent or pointed chin, open-mouth expression, and uplifted earlobes. Four different heterozygous mutations were identified; 2 mutations were frameshift (c.246_247delGGinsC, c.980_980delG), 1 was nonsense (c.2083C>T), and 1 was splice site (c.808-2A>G). Two of them (c.246_247delGGinsC, c.980_980delG) have not been previously reported in the literature. By defining 2 novel mutations, this study contributes to the molecular spectrum of MWS, while also providing a further insight for genetic counseling. It also demonstrates the importance of dysmorphological examination in clinical diagnosis.
Copyright © 2020 by S. Karger AG, Basel.

Entities:  

Keywords:  Facial dysmorphism; Genetic syndrome; Language impairment; Mowat-Wilson syndrome; ZEB2

Year:  2020        PMID: 33510600      PMCID: PMC7802445          DOI: 10.1159/000511609

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  11 in total

1.  ZFHX1B mutations in patients with Mowat-Wilson syndrome.

Authors:  Florence Dastot-Le Moal; Meredith Wilson; David Mowat; Nathalie Collot; Florence Niel; Michel Goossens
Journal:  Hum Mutat       Date:  2007-04       Impact factor: 4.878

Review 2.  Mowat-Wilson syndrome: facial phenotype changing with age: study of 19 Italian patients and review of the literature.

Authors:  L Garavelli; M Zollino; P Cerruti Mainardi; F Gurrieri; F Rivieri; F Soli; R Verri; E Albertini; E Favaron; M Zignani; D Orteschi; P Bianchi; F Faravelli; F Forzano; M Seri; A Wischmeijer; D Turchetti; E Pompilii; M Gnoli; G Cocchi; L Mazzanti; R Bergamaschi; D De Brasi; M P Sperandeo; F Mari; V Uliana; R Mostardini; M Cecconi; M Grasso; S Sassi; G Sebastio; A Renieri; M Silengo; S Bernasconi; N Wakamatsu; G Neri
Journal:  Am J Med Genet A       Date:  2009-03       Impact factor: 2.802

Review 3.  Hirschsprung's disease in children with Mowat-Wilson syndrome.

Authors:  David Coyle; Prem Puri
Journal:  Pediatr Surg Int       Date:  2015-07-09       Impact factor: 1.827

4.  The behavioral phenotype of Mowat-Wilson syndrome.

Authors:  Elizabeth Evans; Stewart Einfeld; David Mowat; John Taffe; Bruce Tonge; Meredith Wilson
Journal:  Am J Med Genet A       Date:  2012-01-13       Impact factor: 2.802

5.  Epilepsy in Mowat-Wilson syndrome: delineation of the electroclinical phenotype.

Authors:  Duccio Maria Cordelli; Livia Garavelli; Salvatore Savasta; Azzurra Guerra; Alessandro Pellicciari; Lucio Giordano; Silvia Bonetti; Ilaria Cecconi; Anita Wischmeijer; Marco Seri; Simonetta Rosato; Chiara Gelmini; Elvio Della Giustina; Anna Rita Ferrari; Nicoletta Zanotta; Roberta Epifanio; Daniele Grioni; Baris Malbora; Isabella Mammi; Francesca Mari; Sabrina Buoni; Rosa Mostardini; Salvatore Grosso; Chiara Pantaleoni; Morena Doz; Maria Luisa Poch-Olivé; Francesca Rivieri; Giovanni Sorge; Graziella Simonte; Francesca Licata; Luigi Tarani; Emanuela Terazzi; Laura Mazzanti; Paola Cerruti Mainardi; Antonella Boni; Francesca Faravelli; Marina Grasso; Paolo Bianchi; Marcella Zollino; Emilio Franzoni
Journal:  Am J Med Genet A       Date:  2013-01-15       Impact factor: 2.802

6.  Comprehensive ZEB2 gene analysis for Mowat-Wilson syndrome in a North American cohort: a suggested approach to molecular diagnostics.

Authors:  Carol J Saunders; Weiwei Zhao; Holly H Ardinger
Journal:  Am J Med Genet A       Date:  2009-11       Impact factor: 2.802

7.  Mowat-Wilson syndrome in a Chinese population: A case series.

Authors:  Stephanie Ho; Ho-Ming Luk; Brian Hon-Yin Chung; Jasmine Lee-Fong Fung; Harriet Hang-Yee Mak; Ivan F M Lo
Journal:  Am J Med Genet A       Date:  2020-03-20       Impact factor: 2.802

8.  A Diagnosis to Consider in Intellectual Disability: Mowat-Wilson Syndrome.

Authors:  Esra Kilic; Arda Cetinkaya; Gülen Eda Utine; Koray Boduroğlu
Journal:  J Child Neurol       Date:  2016-01-25       Impact factor: 1.987

9.  Phenotype and genotype of 87 patients with Mowat-Wilson syndrome and recommendations for care.

Authors:  Ivan Ivanovski; Olivera Djuric; Stefano Giuseppe Caraffi; Daniela Santodirocco; Marzia Pollazzon; Simonetta Rosato; Duccio Maria Cordelli; Ebtesam Abdalla; Patrizia Accorsi; Margaret P Adam; Paola Francesca Ajmone; Magdalena Badura-Stronka; Chiara Baldo; Maddalena Baldi; Allan Bayat; Stefania Bigoni; Federico Bonvicini; Jeroen Breckpot; Bert Callewaert; Guido Cocchi; Goran Cuturilo; Daniele De Brasi; Koenraad Devriendt; Mary Beth Dinulos; Tina Duelund Hjortshøj; Roberta Epifanio; Francesca Faravelli; Agata Fiumara; Debora Formisano; Lucio Giordano; Marina Grasso; Sabine Grønborg; Alessandro Iodice; Lorenzo Iughetti; Vladimir Kuburovic; Anna Kutkowska-Kazmierczak; Didier Lacombe; Caterina Lo Rizzo; Anna Luchetti; Baris Malbora; Isabella Mammi; Francesca Mari; Giulia Montorsi; Sebastien Moutton; Rikke S Møller; Petra Muschke; Jens Erik Klint Nielsen; Ewa Obersztyn; Chiara Pantaleoni; Alessandro Pellicciari; Maria Antonietta Pisanti; Igor Prpic; Maria Luisa Poch-Olive; Federico Raviglione; Alessandra Renieri; Emilia Ricci; Francesca Rivieri; Gijs W Santen; Salvatore Savasta; Gioacchino Scarano; Ina Schanze; Angelo Selicorni; Margherita Silengo; Robert Smigiel; Luigina Spaccini; Giovanni Sorge; Krzysztof Szczaluba; Luigi Tarani; Luis Gonzaga Tone; Annick Toutain; Aurelien Trimouille; Elvis Terci Valera; Samantha Schrier Vergano; Nicoletta Zanotta; Martin Zenker; Andrea Conidi; Marcella Zollino; Anita Rauch; Christiane Zweier; Livia Garavelli
Journal:  Genet Med       Date:  2018-01-04       Impact factor: 8.822

10.  Mowat-Wilson syndrome: growth charts.

Authors:  Ivan Ivanovski; Olivera Djuric; Serena Broccoli; Stefano Giuseppe Caraffi; Patrizia Accorsi; Margaret P Adam; Kristina Avela; Magdalena Badura-Stronka; Allan Bayat; Jill Clayton-Smith; Isabella Cocco; Duccio Maria Cordelli; Goran Cuturilo; Veronica Di Pisa; Juliette Dupont Garcia; Roberto Gastaldi; Lucio Giordano; Andrea Guala; Christina Hoei-Hansen; Mie Inaba; Alessandro Iodice; Jens Erik Klint Nielsen; Vladimir Kuburovic; Brissia Lazalde-Medina; Baris Malbora; Seiji Mizuno; Oana Moldovan; Rikke S Møller; Petra Muschke; Valeria Otelli; Chiara Pantaleoni; Carmelo Piscopo; Maria Luisa Poch-Olive; Igor Prpic; Purificación Marín Reina; Federico Raviglione; Emilia Ricci; Emanuela Scarano; Graziella Simonte; Robert Smigiel; George Tanteles; Luigi Tarani; Aurelien Trimouille; Elvis Terci Valera; Samantha Schrier Vergano; Karin Writzl; Bert Callewaert; Salvatore Savasta; Maria Elisabeth Street; Lorenzo Iughetti; Sergio Bernasconi; Paolo Giorgi Rossi; Livia Garavelli
Journal:  Orphanet J Rare Dis       Date:  2020-06-15       Impact factor: 4.123

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