Literature DB >> 15918153

3q29 microdeletion syndrome: clinical and molecular characterization of a new syndrome.

Lionel Willatt1, James Cox, John Barber, Elisabet Dachs Cabanas, Amanda Collins, Dian Donnai, David R FitzPatrick, Eddy Maher, Howard Martin, Josep Parnau, Lesley Pindar, Jacqueline Ramsay, Charles Shaw-Smith, Erik A Sistermans, Michael Tettenborn, Dorothy Trump, Bert B A de Vries, Kate Walker, F Lucy Raymond.   

Abstract

We report the identification of six patients with 3q29 microdeletion syndrome. The clinical phenotype is variable despite an almost identical deletion size. The phenotype includes mild-to-moderate mental retardation, with only slightly dysmorphic facial features that are similar in most patients: a long and narrow face, short philtrum, and high nasal bridge. Autism, gait ataxia, chest-wall deformity, and long and tapering fingers were noted in at least two of six patients. Additional features--including microcephaly, cleft lip and palate, horseshoe kidney and hypospadias, ligamentous laxity, recurrent middle ear infections, and abnormal pigmentation--were observed, but each feature was only found once, in a single patient. The microdeletion is approximately 1.5 Mb in length, with molecular boundaries mapping within the same or adjacent bacterial artificial chromosome (BAC) clones at either end of the deletion in all patients. The deletion encompasses 22 genes, including PAK2 and DLG1, which are autosomal homologues of two known X-linked mental retardation genes, PAK3 and DLG3. The presence of two nearly identical low-copy repeat sequences in BAC clones on each side of the deletion breakpoint suggests that nonallelic homologous recombination is the likely mechanism of disease causation in this syndrome.

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Year:  2005        PMID: 15918153      PMCID: PMC1226188          DOI: 10.1086/431653

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  20 in total

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Journal:  Nat Genet       Date:  2001-11       Impact factor: 38.330

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Authors:  Heidi A Heilstedt; Blake C Ballif; Leslie A Howard; Richard A Lewis; Samuel Stal; Catherine D Kashork; Carlos A Bacino; Stuart K Shapira; Lisa G Shaffer
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Review 3.  Implications of human genome architecture for rearrangement-based disorders: the genomic basis of disease.

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Review 4.  Telomeres: a diagnosis at the end of the chromosomes.

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Journal:  J Med Genet       Date:  2003-06       Impact factor: 6.318

5.  Molecular analysis of 20 patients with 2q37.3 monosomy: definition of minimum deletion intervals for key phenotypes.

Authors:  M A Aldred; R O C Sanford; N S Thomas; M A Barrow; L C Wilson; L A Brueton; M C Bonaglia; R C M Hennekam; C Eng; N R Dennis; R C Trembath
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6.  Quaternary structure, protein dynamics, and synaptic function of SAP97 controlled by L27 domain interactions.

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7.  Screening for subtelomeric rearrangements in 210 patients with unexplained mental retardation using multiplex ligation dependent probe amplification (MLPA).

Authors:  D A Koolen; W M Nillesen; M H A Versteeg; G F M Merkx; N V A M Knoers; M Kets; S Vermeer; C M A van Ravenswaaij; C G de Kovel; H G Brunner; D Smeets; B B A de Vries; E A Sistermans
Journal:  J Med Genet       Date:  2004-12       Impact factor: 6.318

8.  Subtle chromosomal rearrangements in children with unexplained mental retardation.

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9.  The use of telomere probes to investigate submicroscopic rearrangements associated with mental retardation.

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Journal:  Curr Opin Genet Dev       Date:  2003-06       Impact factor: 5.578

10.  Mutations in the DLG3 gene cause nonsyndromic X-linked mental retardation.

Authors:  Patrick Tarpey; Josep Parnau; Matthew Blow; Hayley Woffendin; Graham Bignell; Charles Cox; James Cox; Helen Davies; Sarah Edkins; Simon Holden; Angelique Korny; Uma Mallya; Jenny Moon; Sarah O'Meara; Adrian Parker; Philip Stephens; Claire Stevens; Jon Teague; Andrew Donnelly; Marie Mangelsdorf; John Mulley; Michael Partington; Gillian Turner; Roger Stevenson; Charles Schwartz; Ian Young; Douglas Easton; Martin Bobrow; P Andrew Futreal; Michael R Stratton; Jozef Gecz; Richard Wooster; F Lucy Raymond
Journal:  Am J Hum Genet       Date:  2004-06-07       Impact factor: 11.025

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  83 in total

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Journal:  J Neurosci       Date:  2011-11-09       Impact factor: 6.167

Review 2.  Microdeletion and microduplication syndromes.

Authors:  Anja Weise; Kristin Mrasek; Elisabeth Klein; Milene Mulatinho; Juan C Llerena; David Hardekopf; Sona Pekova; Samarth Bhatt; Nadezda Kosyakova; Thomas Liehr
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3.  Recurrent deletions and reciprocal duplications of 10q11.21q11.23 including CHAT and SLC18A3 are likely mediated by complex low-copy repeats.

Authors:  Paweł Stankiewicz; Shashikant Kulkarni; Avinash V Dharmadhikari; Srirangan Sampath; Samarth S Bhatt; Tamim H Shaikh; Zhilian Xia; Amber N Pursley; M Lance Cooper; Marwan Shinawi; Alex R Paciorkowski; Dorothy K Grange; Michael J Noetzel; Scott Saunders; Paul Simons; Marshall Summar; Brendan Lee; Fernando Scaglia; Florence Fellmann; Danielle Martinet; Jacques S Beckmann; Alexander Asamoah; Kathryn Platky; Susan Sparks; Ann S Martin; Suneeta Madan-Khetarpal; Jacqueline Hoover; Livija Medne; Carsten G Bonnemann; John B Moeschler; Stephanie E Vallee; Sumit Parikh; Polly Irwin; Victoria P Dalzell; Wendy E Smith; Valerie C Banks; David B Flannery; Carolyn M Lovell; Gary A Bellus; Kathryn Golden-Grant; Jerome L Gorski; Jennifer L Kussmann; Tracy L McGregor; Rizwan Hamid; Jean Pfotenhauer; Blake C Ballif; Chad A Shaw; Sung-Hae L Kang; Carlos A Bacino; Ankita Patel; Jill A Rosenfeld; Sau Wai Cheung; Lisa G Shaffer
Journal:  Hum Mutat       Date:  2011-11-02       Impact factor: 4.878

4.  A case of 3q29 microdeletion syndrome involving oral cleft inherited from a nonaffected mosaic parent: molecular analysis and ethical implications.

Authors:  Aline L Petrin; Sandra Daack-Hirsch; Jamie L'Heureux; Jeffrey C Murray
Journal:  Cleft Palate Craniofac J       Date:  2010-05-04

5.  Segmental duplications mediate novel, clinically relevant chromosome rearrangements.

Authors:  M Katharine Rudd; Julia Keene; Brian Bunke; Erin B Kaminsky; Margaret P Adam; Jennifer G Mulle; David H Ledbetter; Christa L Martin
Journal:  Hum Mol Genet       Date:  2009-05-14       Impact factor: 6.150

6.  Further delineation of the 15q13 microdeletion and duplication syndromes: a clinical spectrum varying from non-pathogenic to a severe outcome.

Authors:  B W M van Bon; H C Mefford; B Menten; D A Koolen; A J Sharp; W M Nillesen; J W Innis; T J L de Ravel; C L Mercer; M Fichera; H Stewart; L E Connell; K Ounap; K Lachlan; B Castle; N Van der Aa; C van Ravenswaaij; M A Nobrega; C Serra-Juhé; I Simonic; N de Leeuw; R Pfundt; E M Bongers; C Baker; P Finnemore; S Huang; V K Maloney; J A Crolla; M van Kalmthout; M Elia; G Vandeweyer; J P Fryns; S Janssens; N Foulds; S Reitano; K Smith; S Parkel; B Loeys; C G Woods; A Oostra; F Speleman; A C Pereira; A Kurg; L Willatt; S J L Knight; J R Vermeesch; C Romano; J C Barber; G Mortier; L A Pérez-Jurado; F Kooy; H G Brunner; E E Eichler; T Kleefstra; B B A de Vries
Journal:  J Med Genet       Date:  2009-04-15       Impact factor: 6.318

7.  Molecular and clinical characterization of de novo and familial cases with microduplication 3q29: guidelines for copy number variation case reporting.

Authors:  S Goobie; J Knijnenburg; D Fitzpatrick; F H Sharkey; A C Lionel; C R Marshall; T Azam; M Shago; K Chong; R Mendoza-Londono; N S den Hollander; C Ruivenkamp; E Maher; H J Tanke; K Szuhai; R F Wintle; S W Scherer
Journal:  Cytogenet Genome Res       Date:  2009-03-11       Impact factor: 1.636

8.  Population analysis of large copy number variants and hotspots of human genetic disease.

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Journal:  Am J Hum Genet       Date:  2009-01-22       Impact factor: 11.025

9.  Ohnologs are overrepresented in pathogenic copy number mutations.

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Review 10.  The genetics of Autism Spectrum Disorders--a guide for clinicians.

Authors:  Karsten M Heil; Christian P Schaaf
Journal:  Curr Psychiatry Rep       Date:  2013-01       Impact factor: 5.285

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