Literature DB >> 26437074

First fetal case of the 8q24.3 contiguous genes syndrome.

Constance Wells1, Emmanuel Spaggiari1,2,3, Valérie Malan1,3, Julien J Stirnemann2,3, Tania Attie-Bitach1,3, Yves Ville2,3, Michel Vekemans1,3, Bettina Bessieres1, Serge Romana1,3.   

Abstract

Molecular cytogenetics, particularly array-CGH, opened the way to the « genotype first approach » and for the discovery of new micro rearrangement syndromes. This was the case for the 8q24.3 microdeletion syndrome. Here, we describe the phenotype of a fetus with a 8q24.3 deletion. This rare condition has to be considered as a contiguous genes syndrome because its phenotype is generated by the SCRIB and PUF60 adjacent gene endophenotypes. The fetus presented atrioventricular septal defect and hypoplastic aortic arch, facial dysmorphism, microretrognathia, dysmorphic ears, clinodactyly of the 5th digit on both hands, mild rocker bottom feet and abnormal third sacral vertebra. This fetus is the first case where the endophenotype produced by SCRIB gene is absent. This case is compared with the previous published cases.
© 2015 Wiley Periodicals, Inc.

Entities:  

Keywords:  PUF60; SCRIB; del 8q24; prenatal array-CGH

Mesh:

Substances:

Year:  2015        PMID: 26437074     DOI: 10.1002/ajmg.a.37411

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  5 in total

1.  Dominant variants in the splicing factor PUF60 cause a recognizable syndrome with intellectual disability, heart defects and short stature.

Authors:  Salima El Chehadeh; Wilhelmina S Kerstjens-Frederikse; Julien Thevenon; Paul Kuentz; Ange-Line Bruel; Christel Thauvin-Robinet; Candace Bensignor; Hélène Dollfus; Vincent Laugel; Jean-Baptiste Rivière; Yannis Duffourd; Caroline Bonnet; Matthieu P Robert; Rodica Isaiko; Morgane Straub; Catherine Creuzot-Garcher; Patrick Calvas; Nicolas Chassaing; Bart Loeys; Edwin Reyniers; Geert Vandeweyer; Frank Kooy; Miroslava Hančárová; Marketa Havlovicová; Darina Prchalová; Zdenek Sedláček; Christian Gilissen; Rolph Pfundt; Jolien S Klein Wassink-Ruiter; Laurence Faivre
Journal:  Eur J Hum Genet       Date:  2016-11-02       Impact factor: 4.246

2.  Identification of a New Variant of PUF60 Gene: Case Presentation and Literature Review.

Authors:  Daniela Oana Toader; Nicolae Bacalbasa; Radu Ursu; Dragos Cretoiu; Lucian G Pop; Irina Balescu; Florentina Gherghiceanu; Florentina Furtunescu; Daniel Radavoi; Viorica Radoi
Journal:  Cancer Diagn Progn       Date:  2021-07-03

3.  Neuron-Specific Deletion of Scrib in Mice Leads to Neuroanatomical and Locomotor Deficits.

Authors:  Jerome Ezan; Maité M Moreau; Tamrat M Mamo; Miki Shimbo; Maureen Decroo; Nathalie Sans; Mireille Montcouquiol
Journal:  Front Genet       Date:  2022-05-25       Impact factor: 4.772

4.  PUF60 variants cause a syndrome of ID, short stature, microcephaly, coloboma, craniofacial, cardiac, renal and spinal features.

Authors:  Karen J Low; Morad Ansari; Rami Abou Jamra; Angus Clarke; Salima El Chehadeh; David R FitzPatrick; Mark Greenslade; Alex Henderson; Jane Hurst; Kory Keller; Paul Kuentz; Trine Prescott; Franziska Roessler; Kaja K Selmer; Michael C Schneider; Fiona Stewart; Katrina Tatton-Brown; Julien Thevenon; Magnus D Vigeland; Julie Vogt; Marjolaine Willems; Jonathan Zonana; D D D Study; Sarah F Smithson
Journal:  Eur J Hum Genet       Date:  2017-03-22       Impact factor: 4.246

5.  Unraveling the mechanism of recognition of the 3' splice site of the adenovirus major late promoter intron by the alternative splicing factor PUF60.

Authors:  Hsin-Hao T Hsiao; Gregg V Crichlow; James W Murphy; Ewa J Folta-Stogniew; Elias J Lolis; Demetrios T Braddock
Journal:  PLoS One       Date:  2020-11-30       Impact factor: 3.240

  5 in total

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