Literature DB >> 31411728

Meckel syndrome: Clinical and mutation profile in six fetuses.

Periyasamy Radhakrishnan1, Shalini S Nayak1, Anju Shukla1, Anna Lindstrand2, Katta M Girisha1.   

Abstract

Meckel syndrome (MKS) is a perinatally lethal, genetically heterogeneous, autosomal recessive condition caused by defective primary cilium formation leading to polydactyly, multiple cysts in kidneys and malformations of nervous system. We performed exome sequencing in six fetuses from six unrelated families with MKS. We identified seven novel variants in B9D2, TNXDC15, CC2D2A, CEP290 and TMEM67. We describe the second family with MKS due to a homozygous variant in B9D2 and fifth family with bi-allelic variant in TXNDC15. Our data validates the causation of MKS by pathogenic variation in B9D2 and TXNDC15 and also adds novel variants in CC2D2A, CEP290 and TMEM67 to the literature.
© 2019 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  zzm321990B9D2; zzm321990CC2D2A; zzm321990CEP290; zzm321990TMEM67; zzm321990TXNDC15; Meckel syndrome; cystic kidneys; holoprosencephaly; polydactyly

Mesh:

Year:  2019        PMID: 31411728     DOI: 10.1111/cge.13623

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


  6 in total

Review 1.  Introducing Thioredoxin-Related Transmembrane Proteins: Emerging Roles of Human TMX and Clinical Implications.

Authors:  Yoshiyuki Matsuo
Journal:  Antioxid Redox Signal       Date:  2021-12-07       Impact factor: 7.468

2.  Two novel TCTN2 mutations cause Meckel-Gruber syndrome.

Authors:  Manli Zhang; Zhijie Chang; Yaping Tian; Longxia Wang; Yanping Lu
Journal:  J Hum Genet       Date:  2020-07-12       Impact factor: 3.172

3.  Evaluation of novel compound variants of CEP290 in prenatally suspected case of Meckel syndrome through whole exome sequencing.

Authors:  Meilian Peng; Shuai Han; Juan Sun; Xiaodong He; Yaer Lv; Liwei Yang
Journal:  Mol Genet Genomic Med       Date:  2022-03-30       Impact factor: 2.473

4.  Three Novel Variants of CEP290 and CC2D2DA and a Link Between ZNF77 and SHH Signaling Pathway Are Found in Two Meckel-Gruber Syndrome Fetuses.

Authors:  Zhidan Hong; Xuanyi He; Fang Yu; Huanyu Liu; Xiaoli Zhang; Yuanzhen Zhang
Journal:  Reprod Sci       Date:  2022-01-03       Impact factor: 2.924

5.  Novel homozygous nonsense mutation associated with Bardet-Biedl syndrome in fetuses with congenital renal malformation.

Authors:  Meiying Cai; Min Lin; Na Lin; Liangpu Xu; Hailong Huang
Journal:  Medicine (Baltimore)       Date:  2022-08-12       Impact factor: 1.817

Review 6.  Thioredoxin-Related Transmembrane Proteins: TMX1 and Little Brothers TMX2, TMX3, TMX4 and TMX5.

Authors:  Concetta Guerra; Maurizio Molinari
Journal:  Cells       Date:  2020-08-31       Impact factor: 6.600

  6 in total

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