Literature DB >> 28815877

New intragenic rearrangements in non-Finnish mulibrey nanism.

Florence Jobic1,2, Gilles Morin1, Catherine Vincent-Delorme3, Estelle Cadet2, Rosalie Cabry4, Michèle Mathieu-Dramard1, Henri Copin4, Jacques Rochette2, Guillaume Jedraszak4.   

Abstract

Prenatal growth is a complex dynamic process controlled by various genetic and environmental factors. Among genetic syndromes characterized by growth restriction, MULIBREY nanism represents a rare autosomal recessive condition presenting with severe pre- and post-natal growth failure, characteristic dysmorphic features but normal neurological development. The phenotype of MULIBREY nanism is variable and overlaps with others such as the Silver-Russell syndrome. We report here three patients in two distinct non-Finnish families from North France who were first suspected to have Silver-Russell syndrome which failed to be confirmed on molecular analyses. Clinical features in the three patients led us to also consider the diagnosis of MULIBREY nanism. Sequencing of the TRIM37 gene showed the three patients shared a novel nonsense mutation (c.181 C>T p.Arg61*) in a heterozygous state. Quantitative fluorescent multiplex PCR identified a new deletion of exons 15 and 16 in TRIM37 in one isolated patient and another deletion of exon 9 in two siblings. Breakpoints of both the deletions were localized in Alu sequences. Given the high number of Alu repeats, which predispose to gene rearrangements, one should always consider such genetic rearrangements in the molecular diagnosis of non-Finnish MULIBREY nanism patients. Early diagnosis of the disease would prompt careful cardiac follow up of such patients as cardiological complication is a characteristic feature of the MULIBREY nanism as described in this report.
© 2017 Wiley Periodicals, Inc.

Entities:  

Keywords:  cardiopathy; deletion; mulibrey nanism; mutation; quantitative PCR

Mesh:

Substances:

Year:  2017        PMID: 28815877     DOI: 10.1002/ajmg.a.38381

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


  6 in total

Review 1.  Warning SINEs: Alu elements, evolution of the human brain, and the spectrum of neurological disease.

Authors:  Peter A Larsen; Kelsie E Hunnicutt; Roxanne J Larsen; Anne D Yoder; Ann M Saunders
Journal:  Chromosome Res       Date:  2018-02-19       Impact factor: 5.239

2.  Exploring the genetics of trotting racing ability in horses using a unique Nordic horse model.

Authors:  Brandon D Velie; Mette Lillie; Kim Jäderkvist Fegraeus; Maria K Rosengren; Marina Solé; Maja Wiklund; Carl-Fredrik Ihler; Eric Strand; Gabriella Lindgren
Journal:  BMC Genomics       Date:  2019-02-04       Impact factor: 3.969

3.  Molecular Etiology Disclosed by Array CGH in Patients With Silver-Russell Syndrome or Similar Phenotypes.

Authors:  Milena Crippa; Maria Teresa Bonati; Luciano Calzari; Chiara Picinelli; Cristina Gervasini; Alessandra Sironi; Ilaria Bestetti; Sara Guzzetti; Simonetta Bellone; Angelo Selicorni; Alessandro Mussa; Andrea Riccio; Giovanni Battista Ferrero; Silvia Russo; Lidia Larizza; Palma Finelli
Journal:  Front Genet       Date:  2019-10-15       Impact factor: 4.599

4.  The Importance of Early Pericardiectomy in Mulibrey Nanism Syndrome, a Case Report.

Authors:  Andres Cordova Sanchez; Mostafa Vasigh; Robert Carhart
Journal:  J Investig Med High Impact Case Rep       Date:  2022 Jan-Dec

Review 5.  Wilms tumor with Mulibrey Nanism: A case report and review of literature.

Authors:  Karthik Upasana; Dhwanee Thakkar; Dheeraj Gautam; Manvinder Singh Sachdev; Anjali Yadav; Rohit Kapoor; Veena Raghunathan; Maninder Singh Dhaliwal; Kartikeya Bhargava; Sandhya Nair; Jaiprakash Sharma; Neha Rastogi; Satya Prakash Yadav
Journal:  Cancer Rep (Hoboken)       Date:  2021-07-26

6.  CD4+ T Cell Defects in a Mulibrey Patient With Specific TRIM37 Mutations.

Authors:  Sara Bruzzaniti; Emilia Cirillo; Rosaria Prencipe; Giuliana Giardino; Maria Teresa Lepore; Federica Garziano; Francesco Perna; Claudio Procaccini; Luigi Mascolo; Cristina Pagano; Valentina Fattorusso; Enza Mozzillo; Maurizio Bifulco; Giuseppe Matarese; Adriana Franzese; Claudio Pignata; Mario Galgani
Journal:  Front Immunol       Date:  2020-09-18       Impact factor: 7.561

  6 in total

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