Literature DB >> 28407396

A further family of Stromme syndrome carrying CENPF mutation.

Ferda Ozkinay1, Tahir Atik1, Esra Isik1, Zeliha Gormez2, Mahmut Sagiroglu2, Ozlem Atan Sahin3, Nergul Corduk4, Huseyin Onay5.   

Abstract

Stromme syndrome is a rare genetic disorder characterized by microcephaly, anterior ocular chamber anomalies, and "apple peel" type jejunal atresia. Here, we report a Stromme syndrome family with two affected siblings with a homozygous truncating frameshift mutation in CENPF. A 3-month-old girl was hospitalized due to prenatally diagnosed microcephaly, microphthalmia, and dysmorphological features. The history of a previous child with the same findings in addition to "apple peel" intestinal atresia had been noted. Regarding the clinical features of both affected siblings, a diagnosis of Stromme syndrome was established. Exome-sequencing of these two cases showed the homozygous mutation (c.5912_5913insA)/(p.T1974Nfs*9) in CENPF. While confirmation of this gene being responsible for Stromme syndrome was pending our results, Filges et al. reported that CENPF was indeed underlying the reason for Stromme syndrome. This is the second case report identifying CENPF mutation as the cause of Stromme syndrome.
© 2017 Wiley Periodicals, Inc.

Entities:  

Keywords:  Stromme syndrome; centromere protein F; ciliopathy; exome; gene discovery; massively parallel sequencing; microcephaly

Mesh:

Substances:

Year:  2017        PMID: 28407396     DOI: 10.1002/ajmg.a.38173

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


  5 in total

1.  Miro-dependent mitochondrial pool of CENP-F and its farnesylated C-terminal domain are dispensable for normal development in mice.

Authors:  Martin Peterka; Benoît Kornmann
Journal:  PLoS Genet       Date:  2019-03-11       Impact factor: 5.917

Review 2.  The Mitotic Apparatus and Kinetochores in Microcephaly and Neurodevelopmental Diseases.

Authors:  Francesca Degrassi; Michela Damizia; Patrizia Lavia
Journal:  Cells       Date:  2019-12-24       Impact factor: 6.600

3.  Biallelic variants in CENPF causing a phenotype distinct from Strømme syndrome.

Authors:  Gerarda Cappuccio; Simona Brillante; Roberta Tammaro; Michele Pinelli; Margherita Lucia De Bernardi; Maria Grazia Gensini; Emilia K Bijlsma; Tamara T Koopmann; Mariette J V Hoffer; Kimberly McDonald; Laura G Hendon; Sofia Douzgou; Charulata Deshpande; Stefano D'Arrigo; Annalaura Torella; Vincenzo Nigro; Brunella Franco; Nicola Brunetti-Pierri
Journal:  Am J Med Genet C Semin Med Genet       Date:  2022-04-30       Impact factor: 3.359

Review 4.  Clinical Application of Genome and Exome Sequencing as a Diagnostic Tool for Pediatric Patients: a Scoping Review of the Literature.

Authors:  Hadley Stevens Smith; J Michael Swint; Seema R Lalani; Jose-Miguel Yamal; Marcia C de Oliveira Otto; Stephan Castellanos; Amy Taylor; Brendan H Lee; Heidi V Russell
Journal:  Genet Med       Date:  2018-05-14       Impact factor: 8.822

5.  Pathogenic alleles in microtubule, secretory granule and extracellular matrix-related genes in familial keratoconus.

Authors:  Vishal Shinde; Nara Sobreira; Elizabeth S Wohler; George Maiti; Nan Hu; Giuliana Silvestri; Sonia George; Jonathan Jackson; Aravinda Chakravarti; Colin E Willoughby; Shukti Chakravarti
Journal:  Hum Mol Genet       Date:  2021-05-17       Impact factor: 6.150

  5 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.