Literature DB >> 12514365

SHORT syndrome.

Rainer Koenig1, Leticia Brendel, Sigrun Fuchs.   

Abstract

We describe a mother and her son with short stature, progeroid facies, Rieger anomaly, teething delay, and mild developmental retardation, particularly speech delay, which are characteristic features of the SHORT syndrome. An additional sign of all described patients is the slight build with lack of subcutaneous fat. Resistance to insulin was suggested by an oral glucose tolerance test in the mother, whereas the test was normal in the index patient at the age of 2 years 2 months. We review the literature and discuss the name-giving symptoms critically. Five familial cases in different generations, equally affected male and female patients and male-to-male transmission point to an autosomal dominant mode of inheritance.

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Year:  2003        PMID: 12514365     DOI: 10.1097/00019605-200301000-00008

Source DB:  PubMed          Journal:  Clin Dysmorphol        ISSN: 0962-8827            Impact factor:   0.816


  10 in total

1.  Mutations in PIK3R1 cause SHORT syndrome.

Authors:  David A Dyment; Amanda C Smith; Diana Alcantara; Jeremy A Schwartzentruber; Lina Basel-Vanagaite; Cynthia J Curry; I Karen Temple; William Reardon; Sahar Mansour; Mushfequr R Haq; Rodney Gilbert; Ordan J Lehmann; Megan R Vanstone; Chandree L Beaulieu; Jacek Majewski; Dennis E Bulman; Mark O'Driscoll; Kym M Boycott; A Micheil Innes
Journal:  Am J Hum Genet       Date:  2013-06-27       Impact factor: 11.025

2.  SHORT syndrome with partial lipodystrophy due to impaired phosphatidylinositol 3 kinase signaling.

Authors:  Kishan Kumar Chudasama; Jonathon Winnay; Stefan Johansson; Tor Claudi; Rainer König; Ingfrid Haldorsen; Bente Johansson; Ju Rang Woo; Dagfinn Aarskog; Jørn V Sagen; C Ronald Kahn; Anders Molven; Pål Rasmus Njølstad
Journal:  Am J Hum Genet       Date:  2013-06-27       Impact factor: 11.025

3.  PIK3R1 mutations cause syndromic insulin resistance with lipoatrophy.

Authors:  Christel Thauvin-Robinet; Martine Auclair; Laurence Duplomb; Martine Caron-Debarle; Magali Avila; Judith St-Onge; Martine Le Merrer; Bernard Le Luyer; Delphine Héron; Michèle Mathieu-Dramard; Pierre Bitoun; Jean-Michel Petit; Sylvie Odent; Jeanne Amiel; Damien Picot; Virginie Carmignac; Julien Thevenon; Patrick Callier; Martine Laville; Yves Reznik; Cédric Fagour; Marie-Laure Nunes; Jacqueline Capeau; Olivier Lascols; Frédéric Huet; Laurence Faivre; Corinne Vigouroux; Jean-Baptiste Rivière
Journal:  Am J Hum Genet       Date:  2013-06-27       Impact factor: 11.025

Review 4.  Lipodystrophies: disorders of adipose tissue biology.

Authors:  Abhimanyu Garg; Anil K Agarwal
Journal:  Biochim Biophys Acta       Date:  2009-01-07

5.  BMP4 loss-of-function mutations in developmental eye disorders including SHORT syndrome.

Authors:  Linda M Reis; Rebecca C Tyler; Kala F Schilter; Omar Abdul-Rahman; Jeffrey W Innis; Beth A Kozel; Adele S Schneider; Tanya M Bardakjian; Edward J Lose; Donna M Martin; Ulrich Broeckel; Elena V Semina
Journal:  Hum Genet       Date:  2011-02-22       Impact factor: 4.132

6.  Iris Malformation and Anterior Segment Dysgenesis in Mice and Humans With a Mutation in PI 3-Kinase.

Authors:  Marie H Solheim; Allen C Clermont; Jonathon N Winnay; Erlend Hallstensen; Anders Molven; Pål R Njølstad; Eyvind Rødahl; C Ronald Kahn
Journal:  Invest Ophthalmol Vis Sci       Date:  2017-06-01       Impact factor: 4.799

Review 7.  SHORT syndrome in a two-year-old girl - case report.

Authors:  Maria Klatka; Izabela Rysz; Katarzyna Kozyra; Agnieszka Polak; Witold Kołłątaj
Journal:  Ital J Pediatr       Date:  2017-05-04       Impact factor: 2.638

Review 8.  Activated PI3 Kinase Delta Syndrome: From Genetics to Therapy.

Authors:  David Michalovich; Sergey Nejentsev
Journal:  Front Immunol       Date:  2018-02-27       Impact factor: 7.561

Review 9.  SHORT syndrome in two Chinese girls: A case report and review of the literature.

Authors:  Yanhong Zhang; Baolan Ji; Jinsheng Li; Yanying Li; Mei Zhang; Bo Ban
Journal:  Mol Genet Genomic Med       Date:  2020-06-29       Impact factor: 2.183

10.  Exome sequencing identifies a novel mutation in PIK3R1 as the cause of SHORT syndrome.

Authors:  Clea Bárcena; Víctor Quesada; Annachiara De Sandre-Giovannoli; Diana A Puente; Joaquín Fernández-Toral; Sabine Sigaudy; Anwar Baban; Nicolas Lévy; Gloria Velasco; Carlos López-Otín
Journal:  BMC Med Genet       Date:  2014-05-02       Impact factor: 2.103

  10 in total

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