Literature DB >> 22693042

Molecular characterization of Joubert syndrome in Saudi Arabia.

Anas M Alazami1, Muneera J Alshammari, Mustafa A Salih, Fatema Alzahrani, Hadia Hijazi, Mohammed Z Seidahmed, Leen Abu Safieh, Mazhor Aldosary, Arif O Khan, Fowzan S Alkuraya.   

Abstract

Joubert syndrome (JS) is a ciliopathy that is defined primarily by typical cerebellar structural and ocular motility defects. The genetic heterogeneity of this condition is significant with 16 genes identified to date. We have used a combination of autozygome-guided candidate gene mutation analysis and exome sequencing to identify the causative mutation in a series of 12 families. The autozygome approach identified mutations in RPGRIP1L, AHI1, TMEM237, and CEP290, while exome sequencing revealed families with truncating mutations in TCTN1 and C5ORF42. Our study, the largest comprehensive molecular series on JS, provides independent confirmation of the recently reported TCTN1, TMEM237, and C5ORF42 as bona fide JS disease genes, and expands the allelic heterogeneity of this disease.
© 2012 Wiley Periodicals, Inc.

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Year:  2012        PMID: 22693042     DOI: 10.1002/humu.22134

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  27 in total

Review 1.  From Planar Cell Polarity to Ciliogenesis and Back: The Curious Tale of the PPE and CPLANE proteins.

Authors:  Paul N Adler; John B Wallingford
Journal:  Trends Cell Biol       Date:  2017-01-30       Impact factor: 20.808

2.  Joubert syndrome: a model for untangling recessive disorders with extreme genetic heterogeneity.

Authors:  R Bachmann-Gagescu; J C Dempsey; I G Phelps; B J O'Roak; D M Knutzen; T C Rue; G E Ishak; C R Isabella; N Gorden; J Adkins; E A Boyle; N de Lacy; D O'Day; A Alswaid; Radha Ramadevi A; L Lingappa; C Lourenço; L Martorell; À Garcia-Cazorla; H Ozyürek; G Haliloğlu; B Tuysuz; M Topçu; P Chance; M A Parisi; I A Glass; J Shendure; D Doherty
Journal:  J Med Genet       Date:  2015-06-19       Impact factor: 6.318

3.  Three Tctn proteins are functionally conserved in the regulation of neural tube patterning and Gli3 processing but not ciliogenesis and Hedgehog signaling in the mouse.

Authors:  Chengbing Wang; Jia Li; Qing Meng; Baolin Wang
Journal:  Dev Biol       Date:  2017-08-08       Impact factor: 3.582

4.  Joubert syndrome in a neonate: case report with literature review.

Authors:  Haifa A Bin Dahman; Abdul-Hakeem M Bin Mubaireek; Zain H Alhaddad
Journal:  Sudan J Paediatr       Date:  2016

5.  Joubert syndrome: genotyping a Northern European patient cohort.

Authors:  Hester Y Kroes; Glen R Monroe; Bert van der Zwaag; Karen J Duran; Carolien G de Kovel; Mark J van Roosmalen; Magdalena Harakalova; Ies J Nijman; Wigard P Kloosterman; Rachel H Giles; Nine V A M Knoers; Gijs van Haaften
Journal:  Eur J Hum Genet       Date:  2015-04-29       Impact factor: 4.246

Review 6.  Tectonic gene mutations in patients with Joubert syndrome.

Authors:  Peter Huppke; Eike Wegener; Helena Böhrer-Rabel; Hanno J Bolz; Barbara Zoll; Jutta Gärtner; Carsten Bergmann
Journal:  Eur J Hum Genet       Date:  2014-08-13       Impact factor: 4.246

7.  Genomic analysis of Meckel-Gruber syndrome in Arabs reveals marked genetic heterogeneity and novel candidate genes.

Authors:  Ranad Shaheen; Eissa Faqeih; Muneera J Alshammari; Abdulrahman Swaid; Lihadh Al-Gazali; Elham Mardawi; Shinu Ansari; Sameera Sogaty; Mohammed Z Seidahmed; Muhammed I AlMotairi; Chantal Farra; Wesam Kurdi; Shatha Al-Rasheed; Fowzan S Alkuraya
Journal:  Eur J Hum Genet       Date:  2012-11-21       Impact factor: 4.246

Review 8.  The application of next-generation sequencing in the autozygosity mapping of human recessive diseases.

Authors:  Fowzan S Alkuraya
Journal:  Hum Genet       Date:  2013-08-02       Impact factor: 4.132

9.  Bi-allelic Mutations in FAM149B1 Cause Abnormal Primary Cilium and a Range of Ciliopathy Phenotypes in Humans.

Authors:  Ranad Shaheen; Nan Jiang; Fatema Alzahrani; Nour Ewida; Tarfa Al-Sheddi; Eman Alobeid; Damir Musaev; Valentina Stanley; Mais Hashem; Niema Ibrahim; Firdous Abdulwahab; Abduljabbar Alshenqiti; Fatma Mujgan Sonmez; Nadia Saqati; Hamad Alzaidan; Mohammad M Al-Qattan; Futwan Al-Mohanna; Joseph G Gleeson; Fowzan S Alkuraya
Journal:  Am J Hum Genet       Date:  2019-03-21       Impact factor: 11.025

Review 10.  Joubert syndrome: congenital cerebellar ataxia with the molar tooth.

Authors:  Marta Romani; Alessia Micalizzi; Enza Maria Valente
Journal:  Lancet Neurol       Date:  2013-07-17       Impact factor: 44.182

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