Literature DB >> 26679067

Prenatal diagnosis of Nager syndrome in a 12-week-old fetus with a whole gene deletion of SF3B4 by chromosomal microarray.

Ida Charlotte Bay Lund1, Else Marie Vestergaard2, Rikke Christensen2, Niels Uldbjerg3, Naja Becher2.   

Abstract

Less than one hundred cases of the acrofacial dysostosis, Nager syndrome, have been described. The cardinal features of Nager syndrome are micrognathia, midface retrusion and limb malformations, predominately of the radial ray of upper extremities. Within the past three years haploinsufficiency of SF3B4 has been confirmed as the major cause of Nager syndrome. Different loss-of-function point-mutations in SF3B4 have been found in approximately 2/3 of patients diagnosed with Nager syndrome. Whole gene deletions of SF3B4 have also been suggested to be the cause of Nager syndrome in SF3B4 point mutation negative patients. Only four prenatal cases displaying Nager-like features in the 2nd or 3rd trimester which have been genetically confirmed with SF3B4 point-mutation after birth have been described. We report a case of a 12-week-old fetus with micrognathia, malformed wrists, bilateral club foot and short long bones diagnosed prenatally by chromosomal microarray with a de novo 0.4 Mb deletion at chromosome 1q21.2 involving SF3B4. To our knowledge, this is the first report of Nager syndrome caused by a SF3B4 whole gene deletion. The case presented also shows that high-resolution chromosomal microarray in early pregnancy can confirm Nager syndrome caused by SF3B4-deletion prenatally.
Copyright © 2015 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  Acrofacial dysostosis; Chromosomal microarray; Nager syndrome; Prenatal diagnosis; SF3B4

Mesh:

Substances:

Year:  2015        PMID: 26679067     DOI: 10.1016/j.ejmg.2015.12.001

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  5 in total

1.  A synonymous splicing mutation in the SF3B4 gene segregates in a family with highly variable Nager syndrome.

Authors:  Matteo Cassina; Cristina Cerqua; Silvia Rossi; Leonardo Salviati; Alessandro Martini; Maurizio Clementi; Eva Trevisson
Journal:  Eur J Hum Genet       Date:  2016-12-14       Impact factor: 4.246

2.  Clinical application of chromosomal microarray analysis for fetuses with craniofacial malformations.

Authors:  Chenyang Xu; Yanbao Xiang; Xueqin Xu; Lili Zhou; Huanzheng Li; Xueqin Dong; Shaohua Tang
Journal:  Mol Cytogenet       Date:  2020-08-25       Impact factor: 2.009

3.  Molecular mechanisms of hearing loss in Nager syndrome.

Authors:  Santosh Kumar Maharana; Jean-Pierre Saint-Jeannet
Journal:  Dev Biol       Date:  2021-04-14       Impact factor: 3.148

4.  Altered mRNA Splicing, Chondrocyte Gene Expression and Abnormal Skeletal Development due to SF3B4 Mutations in Rodriguez Acrofacial Dysostosis.

Authors:  Felipe Marques; Jessica Tenney; Ivan Duran; Jorge Martin; Lisette Nevarez; Robert Pogue; Deborah Krakow; Daniel H Cohn; Bing Li
Journal:  PLoS Genet       Date:  2016-09-13       Impact factor: 5.917

5.  Broad-spectrum next-generation sequencing-based diagnosis of a case of Nager syndrome.

Authors:  Jue Zhao; Liwei Yang
Journal:  J Clin Lab Anal       Date:  2020-06-14       Impact factor: 2.352

  5 in total

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