Literature DB >> 9719381

Distal 6p deletion syndrome: a report of a case with anterior chamber eye anomaly and review of published reports.

C J Law1, A M Fisher, I K Temple.   

Abstract

We describe a 32 year old male with a distal 6p24.3-->pter deletion. He has specific developmental anomalies of the anterior chamber of the eye and a cleft uvula which is consistent with the recent localisation of genes for iris development and orofacial clefting to distal 6p. In addition he has progressive sensorineural deafness and this may localise a gene for deafness to this region. We conclude that a refined distal 6p deletion syndrome exists and includes a characteristic facial appearance with hypertelorism, downward slanting palpebral fissures, tented mouth, smooth philtrum, palatal malformation, ear anomalies, anterior chamber eye defects, progressive sensorineural deafness, cardiac defects, abdominal herniae, small external genitalia, and motor and speech delay.

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Mesh:

Year:  1998        PMID: 9719381      PMCID: PMC1051400          DOI: 10.1136/jmg.35.8.685

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  14 in total

1.  Terminal deletion 6p23: a case report.

Authors:  M H Kormann-Bortolotto; L M Farah; D Soares; M Corbani; R Müller; A C Adell
Journal:  Am J Med Genet       Date:  1990-12

Review 2.  Partial deletion of chromosome 6p: delineation of the syndrome.

Authors:  C G Palmer; P Bader; M L Slovak; D E Comings; M J Pettenati
Journal:  Am J Med Genet       Date:  1991-05-01

3.  Autosomal dominant Axenfeld-Rieger anomaly maps to 6p25.

Authors:  D B Gould; A J Mears; W G Pearce; M A Walter
Journal:  Am J Hum Genet       Date:  1997-09       Impact factor: 11.025

4.  Two rare cases of 6p partial deletion.

Authors:  S M Jalal; V R Macias; H Roop; F Morgan; P King
Journal:  Clin Genet       Date:  1989-09       Impact factor: 4.438

5.  Tandem Y/6 translocation with partial deletion 6 (p23----pter).

Authors:  P C Kelly; W W Blake; J R Davis
Journal:  Clin Genet       Date:  1989-09       Impact factor: 4.438

6.  Clinical evidence for localisation of HLA proximal of chromosome 6p22.

Authors:  E S Sachs; A J Hoogeboom; M F Niermeijer; G M Schreuder
Journal:  Lancet       Date:  1983-03-19       Impact factor: 79.321

7.  Fluorescence in situ hybridization with human chromosome-specific libraries: detection of trisomy 21 and translocations of chromosome 4.

Authors:  D Pinkel; J Landegent; C Collins; J Fuscoe; R Segraves; J Lucas; J Gray
Journal:  Proc Natl Acad Sci U S A       Date:  1988-12       Impact factor: 11.205

8.  Evidence of a locus for orofacial clefting on human chromosome 6p24 and STS content map of the region.

Authors:  A F Davies; R J Stephens; M G Olavesen; L Heather; M J Dixon; A Magee; F Flinter; J Ragoussis
Journal:  Hum Mol Genet       Date:  1995-01       Impact factor: 6.150

9.  Terminal deletion of 6p: report of a new case.

Authors:  A Plaja; R Vidal; D Soriano; X Bou; T Vendrell; C Mediano; J M Pueyo; X Labraña; E Sarret
Journal:  Ann Genet       Date:  1994

10.  Distal deletion of the short arm of chromosome 6.

Authors:  V L Zurcher; W L Golden; A B Zinn
Journal:  Am J Med Genet       Date:  1990-02
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  10 in total

Review 1.  Telomeres: a diagnosis at the end of the chromosomes.

Authors:  B B A De Vries; R Winter; A Schinzel; C van Ravenswaaij-Arts
Journal:  J Med Genet       Date:  2003-06       Impact factor: 6.318

2.  Schizophrenia in an adult with 6p25 deletion syndrome.

Authors:  O Caluseriu; G Mirza; J Ragoussis; E W C Chow; D MacCrimmon; A S Bassett
Journal:  Am J Med Genet A       Date:  2006-06-01       Impact factor: 2.802

3.  Cerebral White Matter Lesions and Dysmorphisms: Signs Suggestive of 6p25 Deletion Syndrome-Literature Review.

Authors:  Piero Pavone; Simona Domenica Marino; Giovanni Corsello; Martino Ruggieri; Danilo Castellano Chiodo; Silvia Marino; Raffaele Falsaperla
Journal:  J Pediatr Genet       Date:  2019-08-04

4.  Deletions in chromosome 6p22.3-p24.3, including ATXN1, are associated with developmental delay and autism spectrum disorders.

Authors:  Patrícia Bs Celestino-Soper; Cindy Skinner; Richard Schroer; Patricia Eng; Jayant Shenai; Malgorzata Mj Nowaczyk; Deborah Terespolsky; Donna Cushing; Gayle S Patel; Ladonna Immken; Alecia Willis; Joanna Wiszniewska; Reuben Matalon; Jill A Rosenfeld; Roger E Stevenson; Sung-Hae L Kang; Sau Wai Cheung; Arthur L Beaudet; Pawel Stankiewicz
Journal:  Mol Cytogenet       Date:  2012-04-05       Impact factor: 2.009

5.  An ENU-induced mutation in AP-2alpha leads to middle ear and ocular defects in Doarad mice.

Authors:  Nadav Ahituv; Alexandra Erven; Helmut Fuchs; Keren Guy; Ruth Ashery-Padan; Trevor Williams; Martin Hrabe de Angelis; Karen B Avraham; Karen P Steel
Journal:  Mamm Genome       Date:  2004-06       Impact factor: 2.957

6.  Haploinsufficiency and triploinsensitivity of the same 6p25.1p24.3 region in a family.

Authors:  Zhongxia Qi; Linda Jo Bone Jeng; Anne Slavotinek; Jingwei Yu
Journal:  BMC Med Genomics       Date:  2015-07-15       Impact factor: 3.063

7.  CAP2 is a regulator of actin pointed end dynamics and myofibrillogenesis in cardiac muscle.

Authors:  Mert Colpan; Jessika Iwanski; Carol C Gregorio
Journal:  Commun Biol       Date:  2021-03-19

8.  QuantiSNP: an Objective Bayes Hidden-Markov Model to detect and accurately map copy number variation using SNP genotyping data.

Authors:  Stefano Colella; Christopher Yau; Jennifer M Taylor; Ghazala Mirza; Helen Butler; Penny Clouston; Anne S Bassett; Anneke Seller; Christopher C Holmes; Jiannis Ragoussis
Journal:  Nucleic Acids Res       Date:  2007-03-06       Impact factor: 16.971

Review 9.  Phenotypic and molecular assessment of seven patients with 6p25 deletion syndrome: relevance to ocular dysgenesis and hearing impairment.

Authors:  Douglas B Gould; Mohamad S Jaafar; Mark K Addison; Francis Munier; Robert Ritch; Ian M MacDonald; Michael A Walter
Journal:  BMC Med Genet       Date:  2004-06-25       Impact factor: 2.103

10.  CAP2 in cardiac conduction, sudden cardiac death and eye development.

Authors:  Jeffrey Field; Diana Z Ye; Manasi Shinde; Fang Liu; Kurt J Schillinger; MinMin Lu; Tao Wang; Michelle Skettini; Yao Xiong; Angela K Brice; Daniel C Chung; Vickas V Patel
Journal:  Sci Rep       Date:  2015-11-30       Impact factor: 4.379

  10 in total

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