Literature DB >> 18661490

Prader-Willi syndrome: is there a recognizable fetal phenotype?

Nicole Bigi1, Jean-Michel Faure, Christine Coubes, Jacques Puechberty, Geneviève Lefort, Pierre Sarda, Patricia Blanchet.   

Abstract

OBJECTIVES: To determine fetal features, which could lead to the diagnosis of Prader-Willi syndrome (PWS) during pregnancy.
METHODS: We analyze the ultrasound features, genetic studies and pathologic findings in two cases of PWS diagnosed during pregnancy.
RESULTS: In the first case, diminished fetal movement, polyhydramnios and oddly positioned hands and feet suggested PWS. Methylation studies confirmed diagnosis and a deletion was detected in the 15q11-q13 region. In the second case, similar ultrasound findings led to prenatal diagnosis of PWS with an abnormal methylation pattern compatible with uniparental disomy. Both fetuses had a characteristic appearance at 28 and 30 weeks' gestation, which included a peculiar position of hands with flexed wrists and dorsi-extended feet with flexed toes.
CONCLUSIONS: The peculiar position of the extremities combined with diminished fetal movement and polyhydramnios seems to be characteristic and should suggest PWS. Copyright (c) 2008 John Wiley & Sons, Ltd.

Entities:  

Mesh:

Year:  2008        PMID: 18661490     DOI: 10.1002/pd.1973

Source DB:  PubMed          Journal:  Prenat Diagn        ISSN: 0197-3851            Impact factor:   3.050


  8 in total

Review 1.  Cellular and disease functions of the Prader-Willi Syndrome gene MAGEL2.

Authors:  Klementina Fon Tacer; Patrick Ryan Potts
Journal:  Biochem J       Date:  2017-06-16       Impact factor: 3.857

Review 2.  Benefits and limitations of prenatal screening for Prader-Willi syndrome.

Authors:  Merlin G Butler
Journal:  Prenat Diagn       Date:  2016-10-12       Impact factor: 3.050

Review 3.  Proteins and proteases of Prader-Willi syndrome: a comprehensive review and perspectives.

Authors:  Sanjukta Basak; Ajoy Basak
Journal:  Biosci Rep       Date:  2022-06-30       Impact factor: 3.976

4.  Truncating Mutations of MAGEL2, a Gene within the Prader-Willi Locus, Are Responsible for Severe Arthrogryposis.

Authors:  Dan Mejlachowicz; Flora Nolent; Jérome Maluenda; Hanitra Ranjatoelina-Randrianaivo; Fabienne Giuliano; Ivo Gut; Damien Sternberg; Annie Laquerrière; Judith Melki
Journal:  Am J Hum Genet       Date:  2015-09-10       Impact factor: 11.025

5.  Polyhydramnios and abnormal foetal heart rate patterns in a foetus with Prader-Willi syndrome: A case report.

Authors:  Tsuyoshi Murata; Toma Fukuda; Aya Kanno; Hyo Kyozuka; Akiko Yamaguchi; Hiromi Shimizu; Takafumi Watanabe; Keiya Fujimori
Journal:  Case Rep Womens Health       Date:  2020-05-29

6.  The phenotypic spectrum of Schaaf-Yang syndrome: 18 new affected individuals from 14 families.

Authors:  Michael D Fountain; Emmelien Aten; Megan T Cho; Jane Juusola; Magdalena A Walkiewicz; Joseph W Ray; Fan Xia; Yaping Yang; Brett H Graham; Carlos A Bacino; Lorraine Potocki; Arie van Haeringen; Claudia A L Ruivenkamp; Pedro Mancias; Hope Northrup; Mary K Kukolich; Marjan M Weiss; Conny M A van Ravenswaaij-Arts; Inge B Mathijssen; Sebastien Levesque; Naomi Meeks; Jill A Rosenfeld; Danielle Lemke; Ada Hamosh; Suzanne K Lewis; Simone Race; Laura L Stewart; Beverly Hay; Andrea M Lewis; Rita L Guerreiro; Jose T Bras; Marcia P Martins; Gerarda Derksen-Lubsen; Els Peeters; Connie Stumpel; Sander Stegmann; Levinus A Bok; Gijs W E Santen; Christian P Schaaf
Journal:  Genet Med       Date:  2016-05-19       Impact factor: 8.822

7.  Neonatal Features of the Prader-Willi Syndrome; The Case for Making the Diagnosis During the First Week of Life

Authors:  Filiz Mine Çizmecioğlu; Jeremy Huw Jones; Wendy Forsyth Paterson; Sakina Kherra; Mariam Kourime; Ruth McGowan; M. Guftar Shaikh; Malcolm Donaldson
Journal:  J Clin Res Pediatr Endocrinol       Date:  2018-03-19

8.  Schaaf-Yang syndrome overview: Report of 78 individuals.

Authors:  John McCarthy; Philip J Lupo; Erin Kovar; Megan Rech; Bret Bostwick; Daryl Scott; Katerina Kraft; Tony Roscioli; Joel Charrow; Samantha A Schrier Vergano; Edward Lose; Robert Smiegel; Yves Lacassie; Christian P Schaaf
Journal:  Am J Med Genet A       Date:  2018-10-10       Impact factor: 2.802

  8 in total

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