Literature DB >> 29159890

Whole exome sequencing reveals a mutation in ARMC9 as a cause of mental retardation, ptosis, and polydactyly.

Anjana Kar1,2, Shubha R Phadke3, Aneek Das Bhowmik2, Ashwin Dalal1.   

Abstract

Intellectual disability (ID) refers to deficits in mental abilities, social behavior, and motor skills to perform activities of daily living as compared to peers. Numerous genetic and environmental factors may be responsible for ID. We report on elucidation of molecular basis for syndromic ID associated with ptosis, polydactyly, and MRI features suggestive of Joubert syndrome using homozygosity mapping followed by exome sequencing. The analysis revealed a novel synonymous variation p.T293T (c.879G>A) which leads to a splicing defect in ARMC9 gene. The variant is present in conserved region of ARM domain of ARMC9 protein, which is predicted to form a platform for protein interaction. This domain is likely to be altered in patient due to splicing defect caused by this synonymous variation. Our report of variant in ARMC9 Leading to Joubert syndrome phenotype (JS30), elucidates the genetic heterogeneity of Joubert syndrome, and expands the gene list for ciliopathies.
© 2017 Wiley Periodicals, Inc.

Entities:  

Keywords:  ARMC9; exome sequencing; homozygosity mapping; intellectual disability; splice site assay

Mesh:

Substances:

Year:  2017        PMID: 29159890     DOI: 10.1002/ajmg.a.38537

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


  5 in total

1.  Proteins that control the geometry of microtubules at the ends of cilia.

Authors:  Panagiota Louka; Krishna Kumar Vasudevan; Mayukh Guha; Ewa Joachimiak; Dorota Wloga; Raphaël F-X Tomasi; Charles N Baroud; Pascale Dupuis-Williams; Domenico F Galati; Chad G Pearson; Luke M Rice; James J Moresco; John R Yates; Yu-Yang Jiang; Karl Lechtreck; William Dentler; Jacek Gaertig
Journal:  J Cell Biol       Date:  2018-09-14       Impact factor: 10.539

Review 2.  Cilia Distal Domain: Diversity in Evolutionarily Conserved Structures.

Authors:  Helena Soares; Bruno Carmona; Sofia Nolasco; Luís Viseu Melo; João Gonçalves
Journal:  Cells       Date:  2019-02-14       Impact factor: 6.600

3.  Whole Exome Sequencing Identified Novel ARMC9 Variations in Two Cases With Joubert Syndrome.

Authors:  Hao Wang; Guanjun Luo; Wensheng Hu; Jin Mei; Yue Shen; Min Wang; Yuan Tan; Yang Yang; Chao Lu; Yong Zhao; Ming Qi
Journal:  Front Genet       Date:  2022-02-04       Impact factor: 4.599

Review 4.  Genotype-phenotype correlates in Joubert syndrome: A review.

Authors:  Simone Gana; Valentina Serpieri; Enza Maria Valente
Journal:  Am J Med Genet C Semin Med Genet       Date:  2022-03-03       Impact factor: 3.359

5.  A CRISPR-based screen for Hedgehog signaling provides insights into ciliary function and ciliopathies.

Authors:  David K Breslow; Sascha Hoogendoorn; Adam R Kopp; David W Morgens; Brandon K Vu; Margaret C Kennedy; Kyuho Han; Amy Li; Gaelen T Hess; Michael C Bassik; James K Chen; Maxence V Nachury
Journal:  Nat Genet       Date:  2018-02-19       Impact factor: 38.330

  5 in total

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