Literature DB >> 30152198

Silver Russel syndrome in an aboriginal patient from Australia.

Cathryn Poulton1,2, Dimitar Azmanov3, Vanessa Atkinson3, John Beilby3,4, Lisa Ewans5, Dylan Gration6, Lauren Dreyer6, Vinutha Shetty7, Ciara Peake8, Emma McCormack9, Richard Palmer10, Barry Lewis11, Hugh Dawkins12, Stephanie Broley6, Gareth Baynam6,13,14,15.   

Abstract

Silver-Russell syndrome (SRS OMIM 180860) is a rare, albeit well-recognized disorder characterized by severe intrauterine and postnatal growth retardation. It remains a clinical diagnosis with a molecular cause identifiable in approximately 60%-70% of patients. We report a 4-year-old Australian Aboriginal girl who was born at 32 weeks gestation with features strongly suggestive of SRS, after extensive investigation she was referred to our undiagnosed disease program (UDP). Genomic sequencing was performed which identified a heterozygous splice site variant in IGF2 which is predicted to be pathogenic by in-silico studies, paternal allelic origin, de novo status, and RNA studies on fibroblasts. We compare clinical findings with reported patients to add to the knowledge base on IGF2 variants and to promote the engagement of other Australian Aboriginal families in genomic medicine.
© 2018 Wiley Periodicals, Inc.

Entities:  

Keywords:  IGF2; Silver-Russel syndrome; aboriginal; macrocephaly

Mesh:

Substances:

Year:  2018        PMID: 30152198     DOI: 10.1002/ajmg.a.40502

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


  7 in total

1.  Rare De Novo IGF2 Variant on the Paternal Allele in a Patient With Silver-Russell Syndrome.

Authors:  Chun-Ling Xia; Yuan Lyu; Chuang Li; Huan Li; Zhi-Tao Zhang; Shao-Wei Yin; Yan Mao; Wen Li; Ling-Yin Kong; Bo Liang; Hong-Kun Jiang; Jesse Li-Ling; Cai-Xia Liu; Jun Wei
Journal:  Front Genet       Date:  2019-11-15       Impact factor: 4.599

Review 2.  Genomic medicine for undiagnosed diseases.

Authors:  Anastasia L Wise; Teri A Manolio; George A Mensah; Josh F Peterson; Dan M Roden; Cecelia Tamburro; Marc S Williams; Eric D Green
Journal:  Lancet       Date:  2019-08-05       Impact factor: 79.321

3.  Components of IGF-axis in growth disorders: a systematic review and patent landscape report.

Authors:  Amit Singh; Ketan Pajni; Inusha Panigrahi; Navdeep Dhoat; Sabyasachi Senapati; Preeti Khetarpal
Journal:  Endocrine       Date:  2022-05-06       Impact factor: 3.925

4.  Novel mutation points to a hot spot in CDKN1C causing Silver-Russell syndrome.

Authors:  Gerhard Binder; Julian Ziegler; Roland Schweizer; Wisam Habhab; Tobias B Haack; Tilman Heinrich; Thomas Eggermann
Journal:  Clin Epigenetics       Date:  2020-10-19       Impact factor: 6.551

5.  Novel Variant in PLAG1 in a Familial Case with Silver-Russell Syndrome Suspicion.

Authors:  Yerai Vado; Arrate Pereda; Isabel Llano-Rivas; Nerea Gorria-Redondo; Ignacio Díez; Guiomar Perez de Nanclares
Journal:  Genes (Basel)       Date:  2020-12-05       Impact factor: 4.096

6.  Case report: A novel de novo IGF2 missense variant in a Finnish patient with Silver-Russell syndrome.

Authors:  Petra Loid; Marita Lipsanen-Nyman; Sirpa Ala-Mello; Katariina Hannula-Jouppi; Juha Kere; Outi Mäkitie; Mari Muurinen
Journal:  Front Pediatr       Date:  2022-10-04       Impact factor: 3.569

Review 7.  Genetic causes of growth hormone insensitivity beyond GHR.

Authors:  Vivian Hwa; Masanobu Fujimoto; Gaohui Zhu; Wen Gao; Corinne Foley; Meenasri Kumbaji; Ron G Rosenfeld
Journal:  Rev Endocr Metab Disord       Date:  2020-10-08       Impact factor: 6.514

  7 in total

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