Literature DB >> 19034313

Interstitial deletion of 6q25.2-q25.3: a novel microdeletion syndrome associated with microcephaly, developmental delay, dysmorphic features and hearing loss.

Sandesh Chakravarthy Sreenath Nagamani1, Ayelet Erez, Christine Eng, Zhishuo Ou, Craig Chinault, Laura Workman, James Coldwell, Pawel Stankiewicz, Ankita Patel, James R Lupski, Sau Wai Cheung.   

Abstract

Interstitial deletions of 6q are rare. We report a detailed clinical and molecular characterization of four patients with interstitial deletion involving 6q25. All of our patients presented with microcephaly, developmental delay, dysmorphic features and hearing loss, whereas two of them had agenesis of the corpus callosum. We determined the size, extent and genomic content of the deletions using high-density array-comparative genomic hybridization (a-CGH), and found that a common segment spanning 3.52 Mb within the 6q25.2-q25.3 region was deleted in all four cases. We hypothesize that a subset of genes in the commonly deleted region are dosage sensitive and that haploinsufficieny of these genes impairs normal development of the brain and hearing.

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Year:  2008        PMID: 19034313      PMCID: PMC2986272          DOI: 10.1038/ejhg.2008.220

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  29 in total

1.  Long arm deletion of chromosome no. 6 in a mentally retarded boy with multiple physical malformations.

Authors:  J Milosević; P Kalicanin
Journal:  J Ment Defic Res       Date:  1975-06

2.  De novo del(6)(q25) associated with macular degeneration.

Authors:  F Rivas; C Ruiz; H Rivera; M Möller; J I Serrano-Lucas; J M Cantú
Journal:  Ann Genet       Date:  1986

3.  A child with partial monosomy 6q secondary to a maternal direct insertional event.

Authors:  S V Matkins; J E Meyer; A C Berry
Journal:  J Med Genet       Date:  1987-04       Impact factor: 6.318

4.  Report of two cases of distal deletion of the long arm of chromosome 6.

Authors:  C A Stevens; R M Fineman; W R Breg; A B Silken
Journal:  Am J Med Genet       Date:  1988-04

5.  Developmental abnormalities associated with long arm deletion of chromosome No. 6.

Authors:  L Bartoshesky; M B Lewis; H M Pashayan
Journal:  Clin Genet       Date:  1978-01       Impact factor: 4.438

6.  Severe sensory hearing loss in del(6q)-syndrome.

Authors:  Maria Schuster; Jörg Lohscheller; Peter Kummer; Ulrich Eysholdt; Frank Rosanowski
Journal:  Int J Pediatr Otorhinolaryngol       Date:  2003-11       Impact factor: 1.675

7.  A DNA replication mechanism for generating nonrecurrent rearrangements associated with genomic disorders.

Authors:  Jennifer A Lee; Claudia M B Carvalho; James R Lupski
Journal:  Cell       Date:  2007-12-28       Impact factor: 41.582

8.  Localization of a novel autosomal recessive non-syndromic hearing impairment locus (DFNB38) to 6q26-q27 in a consanguineous kindred from Pakistan.

Authors:  Muhammad Ansar; Mohammad Ramzan; Thanh L Pham; Kai Yan; Syed Muhammad Jamal; Sayedul Haque; Wasim Ahmad; Suzanne M Leal
Journal:  Hum Hered       Date:  2003       Impact factor: 0.444

9.  Vestibular defects in head-tilt mice result from mutations in Nox3, encoding an NADPH oxidase.

Authors:  Rainer Paffenholz; Rebecca A Bergstrom; Francesca Pasutto; Philipp Wabnitz; Robert J Munroe; Wolfgang Jagla; Ulrich Heinzmann; Andreas Marquardt; Armin Bareiss; Jürgen Laufs; Andreas Russ; Gabriele Stumm; John C Schimenti; David E Bergstrom
Journal:  Genes Dev       Date:  2004-03-10       Impact factor: 11.361

10.  Bacterial artificial chromosome-emulation oligonucleotide arrays for targeted clinical array-comparative genomic hybridization analyses.

Authors:  Zhishuo Ou; Sung-Hae L Kang; Chad A Shaw; Condie E Carmack; Lisa D White; Ankita Patel; Arthur L Beaudet; Sau Wai Cheung; A Craig Chinault
Journal:  Genet Med       Date:  2008-04       Impact factor: 8.822

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  19 in total

1.  Haploinsufficiency of ARID1B, a member of the SWI/SNF-a chromatin-remodeling complex, is a frequent cause of intellectual disability.

Authors:  Juliane Hoyer; Arif B Ekici; Sabine Endele; Bernt Popp; Christiane Zweier; Antje Wiesener; Eva Wohlleber; Andreas Dufke; Eva Rossier; Corinna Petsch; Markus Zweier; Ina Göhring; Alexander M Zink; Gudrun Rappold; Evelin Schröck; Dagmar Wieczorek; Olaf Riess; Hartmut Engels; Anita Rauch; André Reis
Journal:  Am J Hum Genet       Date:  2012-03-09       Impact factor: 11.025

2.  Coffin-Siris Syndrome with obesity, macrocephaly, hepatomegaly and hyperinsulinism caused by a mutation in the ARID1B gene.

Authors:  Mari-Anne Vals; Eve Õiglane-Shlik; Margit Nõukas; Riina Shor; Aleksandr Peet; Mart Kals; Paula Ann Kivistik; Andres Metspalu; Katrin Õunap
Journal:  Eur J Hum Genet       Date:  2014-02-26       Impact factor: 4.246

3.  Necrotizing Enterocolitis in Two Siblings and an Unrelated Infant with Overlapping Chromosome 6q25 Deletions.

Authors:  Hannah C D Esdal; Muhammad B Ghbeis; Daniel A Saltzman; Donavon Hess; Janet R Hume; Robyn C Reed; Susan A Berry; Eric Hoggard; Betsy Hirsch; Linda B Baughn; Lisa A Schimmenti
Journal:  Mol Syndromol       Date:  2018-04-28

4.  Genetic interactions within inositol-related pathways are associated with longitudinal changes in ventricle size.

Authors:  Mary Ellen I Koran; Timothy J Hohman; Shashwath A Meda; Tricia A Thornton-Wells
Journal:  J Alzheimers Dis       Date:  2014       Impact factor: 4.472

5.  Mutations in chromatin regulators functionally link Cornelia de Lange syndrome and clinically overlapping phenotypes.

Authors:  Ilaria Parenti; María E Teresa-Rodrigo; Jelena Pozojevic; Sara Ruiz Gil; Ingrid Bader; Diana Braunholz; Nuria C Bramswig; Cristina Gervasini; Lidia Larizza; Lutz Pfeiffer; Ferda Ozkinay; Feliciano Ramos; Benedikt Reiz; Olaf Rittinger; Tim M Strom; Erwan Watrin; Kerstin Wendt; Dagmar Wieczorek; Bernd Wollnik; Carolina Baquero-Montoya; Juan Pié; Matthew A Deardorff; Gabriele Gillessen-Kaesbach; Frank J Kaiser
Journal:  Hum Genet       Date:  2017-01-24       Impact factor: 4.132

6.  Haploinsufficiency of TAB2 causes congenital heart defects in humans.

Authors:  Bernard Thienpont; Litu Zhang; Alex V Postma; Jeroen Breckpot; Léon-Charles Tranchevent; Peter Van Loo; Kjeld Møllgård; Niels Tommerup; Iben Bache; Zeynep Tümer; Klaartje van Engelen; Björn Menten; Geert Mortier; Darrel Waggoner; Marc Gewillig; Yves Moreau; Koen Devriendt; Lars Allan Larsen
Journal:  Am J Hum Genet       Date:  2010-05-20       Impact factor: 11.025

Review 7.  ARID1B-mediated disorders: Mutations and possible mechanisms.

Authors:  Joe C H Sim; Susan M White; Paul J Lockhart
Journal:  Intractable Rare Dis Res       Date:  2015-02

8.  Cytogenomic delineation and clinical follow-up of two siblings with an 8.5 Mb 6q24.2-q25.2 deletion inherited from a paternal insertion.

Authors:  Vera Ayres Meloni; Roberta Santos Guilherme; Mariana Moyses Oliveira; Michele Migliavacca; Sylvia Satomi Takeno; Nara Lygia Macena Sobreira; Maria de Fatima Faria Soares; Claudia Berlim de Mello; Maria Isabel Melaragno
Journal:  Am J Med Genet A       Date:  2014-06-04       Impact factor: 2.802

9.  The role of BAF (mSWI/SNF) complexes in mammalian neural development.

Authors:  Esther Y Son; Gerald R Crabtree
Journal:  Am J Med Genet C Semin Med Genet       Date:  2014-09-05       Impact factor: 3.908

10.  Evolutionary conserved longevity genes and human cognitive abilities in elderly cohorts.

Authors:  Lorna M Lopez; Sarah E Harris; Michelle Luciano; Dave Liewald; Gail Davies; Alan J Gow; Albert Tenesa; Antony Payton; Xiayi Ke; Lawrence J Whalley; Helen Fox; Paul Haggerty; William Ollier; Andrew Pickles; David J Porteous; Michael A Horan; Neil Pendleton; John M Starr; Ian J Deary
Journal:  Eur J Hum Genet       Date:  2011-11-02       Impact factor: 4.246

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