Literature DB >> 33386993

PIGF deficiency causes a phenotype overlapping with DOORS syndrome.

Smrithi Salian1, Hind Benkerroum1, Thi Tuyet Mai Nguyen1, Sheela Nampoothiri2, Taroh Kinoshita3, Têmis Maria Félix4, Fiona Stewart5, Sanjay M Sisodiya6, Yoshiko Murakami3, Philippe M Campeau7.   

Abstract

DOORS syndrome is characterized by deafness, onychodystrophy, osteodystrophy, intellectual disability, and seizures. In this study, we report two unrelated individuals with DOORS syndrome without deafness. Exome sequencing revealed a homozygous missense variant in PIGF (NM_173074.3:c.515C>G, p.Pro172Arg) in both. We demonstrate impaired glycosylphosphatidylinositol (GPI) biosynthesis through flow cytometry analysis. We thus describe the causal role of a novel disease gene, PIGF, in DOORS syndrome and highlight the overlap between this condition and GPI deficiency disorders. For each gene implicated in DOORS syndrome and/or inherited GPI deficiencies, there is considerable clinical variability so a high index of suspicion is warranted even though not all features are noted.

Entities:  

Year:  2021        PMID: 33386993     DOI: 10.1007/s00439-020-02251-2

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  12 in total

1.  Prediction of membrane-protein topology from first principles.

Authors:  Andreas Bernsel; Håkan Viklund; Jenny Falk; Erik Lindahl; Gunnar von Heijne; Arne Elofsson
Journal:  Proc Natl Acad Sci U S A       Date:  2008-05-13       Impact factor: 11.205

Review 2.  The glypican family of heparan sulfate proteoglycans: major cell-surface proteoglycans of the developing nervous system.

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Journal:  Perspect Dev Neurobiol       Date:  1996

Review 3.  Clinical variability in inherited glycosylphosphatidylinositol deficiency disorders.

Authors:  Kara Bellai-Dussault; Thi Tuyet Mai Nguyen; Nissan V Baratang; Daniel A Jimenez-Cruz; Philippe M Campeau
Journal:  Clin Genet       Date:  2018-08-16       Impact factor: 4.438

4.  Requirement of PIG-F and PIG-O for transferring phosphoethanolamine to the third mannose in glycosylphosphatidylinositol.

Authors:  Y Hong; Y Maeda; R Watanabe; N Inoue; K Ohishi; T Kinoshita
Journal:  J Biol Chem       Date:  2000-07-07       Impact factor: 5.157

5.  GPI7 is the second partner of PIG-F and involved in modification of glycosylphosphatidylinositol.

Authors:  Nobue Shishioh; Yeongjin Hong; Kazuhito Ohishi; Hisashi Ashida; Yusuke Maeda; Taroh Kinoshita
Journal:  J Biol Chem       Date:  2005-01-04       Impact factor: 5.157

6.  Recessive loss of function PIGN alleles, including an intragenic deletion with founder effect in La Réunion Island, in patients with Fryns syndrome.

Authors:  Jean-Luc Alessandri; Christopher T Gordon; Marie-Line Jacquemont; Nicolas Gruchy; Norbert F Ajeawung; Guillaume Benoist; Myriam Oufadem; Asma Chebil; Yannis Duffourd; Coralie Dumont; Marion Gérard; Paul Kuentz; Thibaud Jouan; Francesca Filippini; Thi Tuyet Mai Nguyen; Olivier Alibeu; Christine Bole-Feysot; Patrick Nitschké; Asma Omarjee; Duksha Ramful; Hanitra Randrianaivo; Bérénice Doray; Laurence Faivre; Jeanne Amiel; Philippe M Campeau; Julien Thevenon
Journal:  Eur J Hum Genet       Date:  2018-01-12       Impact factor: 4.246

7.  New insights into the functions of PIGF, a protein involved in the ethanolamine phosphate transfer steps of glycosylphosphatidylinositol biosynthesis.

Authors:  Matthew J Stokes; Yoshiko Murakami; Yusuke Maeda; Taroh Kinoshita; Yasu S Morita
Journal:  Biochem J       Date:  2014-10-15       Impact factor: 3.857

8.  The genetic basis of DOORS syndrome: an exome-sequencing study.

Authors:  Philippe M Campeau; Dalia Kasperaviciute; James T Lu; Lindsay C Burrage; Choel Kim; Mutsuki Hori; Berkley R Powell; Fiona Stewart; Têmis Maria Félix; Jenneke van den Ende; Marzena Wisniewska; Hülya Kayserili; Patrick Rump; Sheela Nampoothiri; Salim Aftimos; Antje Mey; Lal D V Nair; Michael L Begleiter; Isabelle De Bie; Girish Meenakshi; Mitzi L Murray; Gabriela M Repetto; Mahin Golabi; Edward Blair; Alison Male; Fabienne Giuliano; Ariana Kariminejad; William G Newman; Sanjeev S Bhaskar; Jonathan E Dickerson; Bronwyn Kerr; Siddharth Banka; Jacques C Giltay; Dagmar Wieczorek; Anna Tostevin; Joanna Wiszniewska; Sau Wai Cheung; Raoul C Hennekam; Richard A Gibbs; Brendan H Lee; Sanjay M Sisodiya
Journal:  Lancet Neurol       Date:  2013-11-29       Impact factor: 44.182

9.  TBC1D24 genotype-phenotype correlation: Epilepsies and other neurologic features.

Authors:  Simona Balestrini; Mathieu Milh; Claudia Castiglioni; Kevin Lüthy; Mattea J Finelli; Patrik Verstreken; Aaron Cardon; Barbara Gnidovec Stražišar; J Lloyd Holder; Gaetan Lesca; Maria M Mancardi; Anne L Poulat; Gabriela M Repetto; Siddharth Banka; Leonilda Bilo; Laura E Birkeland; Friedrich Bosch; Knut Brockmann; J Helen Cross; Diane Doummar; Temis M Félix; Fabienne Giuliano; Mutsuki Hori; Irina Hüning; Hulia Kayserili; Usha Kini; Melissa M Lees; Girish Meenakshi; Leena Mewasingh; Alistair T Pagnamenta; Silvio Peluso; Antje Mey; Gregory M Rice; Jill A Rosenfeld; Jenny C Taylor; Matthew M Troester; Christine M Stanley; Dorothee Ville; Magdalena Walkiewicz; Antonio Falace; Anna Fassio; Johannes R Lemke; Saskia Biskup; Jessica Tardif; Norbert F Ajeawung; Aslihan Tolun; Mark Corbett; Jozef Gecz; Zaid Afawi; Katherine B Howell; Karen L Oliver; Samuel F Berkovic; Ingrid E Scheffer; Fabrizio A de Falco; Peter L Oliver; Pasquale Striano; Federico Zara; Phillipe M Campeau; S M Sisodiya
Journal:  Neurology       Date:  2016-06-08       Impact factor: 9.910

10.  The mutational constraint spectrum quantified from variation in 141,456 humans.

Authors:  Konrad J Karczewski; Laurent C Francioli; Grace Tiao; Beryl B Cummings; Jessica Alföldi; Qingbo Wang; Ryan L Collins; Kristen M Laricchia; Andrea Ganna; Daniel P Birnbaum; Laura D Gauthier; Harrison Brand; Matthew Solomonson; Nicholas A Watts; Daniel Rhodes; Moriel Singer-Berk; Eleina M England; Eleanor G Seaby; Jack A Kosmicki; Raymond K Walters; Katherine Tashman; Yossi Farjoun; Eric Banks; Timothy Poterba; Arcturus Wang; Cotton Seed; Nicola Whiffin; Jessica X Chong; Kaitlin E Samocha; Emma Pierce-Hoffman; Zachary Zappala; Anne H O'Donnell-Luria; Eric Vallabh Minikel; Ben Weisburd; Monkol Lek; James S Ware; Christopher Vittal; Irina M Armean; Louis Bergelson; Kristian Cibulskis; Kristen M Connolly; Miguel Covarrubias; Stacey Donnelly; Steven Ferriera; Stacey Gabriel; Jeff Gentry; Namrata Gupta; Thibault Jeandet; Diane Kaplan; Christopher Llanwarne; Ruchi Munshi; Sam Novod; Nikelle Petrillo; David Roazen; Valentin Ruano-Rubio; Andrea Saltzman; Molly Schleicher; Jose Soto; Kathleen Tibbetts; Charlotte Tolonen; Gordon Wade; Michael E Talkowski; Benjamin M Neale; Mark J Daly; Daniel G MacArthur
Journal:  Nature       Date:  2020-05-27       Impact factor: 69.504

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