Literature DB >> 17512813

A 2.6 Mb deletion of 6q24.3-25.1 in a patient with growth failure, cardiac septal defect, thin upperlip and asymmetric dysmorphic ears.

R Caselli1, M A Mencarelli, F T Papa, V Uliana, S Schiavone, M Strambi, C Pescucci, F Ariani, V Rossi, I Longo, I Meloni, A Renieri, F Mari.   

Abstract

We report a female patient with neurodevelopmental delay and peculiar facial features. She has postnatal growth failure and an atrial septal defect. Patent duct arteriosis and tricuspidal insufficiency were also noted at birth. Characteristic facial features include medial flare eyebrows, dysmorphic helix of the right ear, cupshaped left ear, anteverted nares, long and smooth philtrum, thin upper lip, high vaulted palate. Array-CGH analysis demonstrated the presence of a 2.6 Mb deletion in 6q24.3-25.1. The phenotypic features of this case are very similar to those previously reported in a patient with a 7Mb overlapping deletion, pointing to a specific new syndrome. Twenty-two genes are present in the common critical deleted region. Among them, there is the PPP1R14C gene that encodes for KEPI, a PKC-potentiated inhibitory protein for type-1 Ser/Thr protein phosphatase. Its selective distribution in brain and heart well correlates with developmental delay and cardiac anomalies observed in the patient.

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Year:  2007        PMID: 17512813     DOI: 10.1016/j.ejmg.2007.03.003

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  7 in total

1.  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

2.  TAB2 deletions and variants cause a highly recognisable syndrome with mitral valve disease, cardiomyopathy, short stature and hypermobility.

Authors:  Aafke Engwerda; Erika K S M Leenders; Barbara Frentz; Paulien A Terhal; Katharina Löhner; Bert B A de Vries; Trijnie Dijkhuizen; Yvonne J Vos; Tuula Rinne; Maarten P van den Berg; Marc T R Roofthooft; Patrick Deelen; Conny M A van Ravenswaaij-Arts; Wilhelmina S Kerstjens-Frederikse
Journal:  Eur J Hum Genet       Date:  2021-08-30       Impact factor: 4.246

3.  Genomic imbalances in neonates with birth defects: high detection rates by using chromosomal microarray analysis.

Authors:  Xin-Yan Lu; Mai T Phung; Chad A Shaw; Kim Pham; Sarah E Neil; Ankita Patel; Trilochan Sahoo; Carlos A Bacino; Pawel Stankiewicz; Sung-Hae Lee Kang; Seema Lalani; A Craig Chinault; James R Lupski; Sau W Cheung; Arthur L Beaudet
Journal:  Pediatrics       Date:  2008-12       Impact factor: 7.124

4.  Whole-exome sequencing and whole genome re-sequencing for prenatal diagnosis of achondroplasia.

Authors:  Rong Zhao; Yan Ruan; Xin Wang
Journal:  Int J Clin Exp Med       Date:  2015-10-15

5.  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

6.  Dysmorphic features and congenital heart disease in chromosome 6q deletion: A short report.

Authors:  Sreelata Nair; Rini Varghese; Sajeed Hashim; Pappachan Scariah
Journal:  Indian J Hum Genet       Date:  2012-01

Review 7.  A new case of de novo 6q24.2-q25.2 deletion on paternal chromosome 6 with growth hormone deficiency: a twelve-year follow-up and literature review.

Authors:  Stefano Stagi; Elisabetta Lapi; Marilena Pantaleo; Massimo Carella; Antonio Petracca; Agostina De Crescenzo; Leopoldo Zelante; Andrea Riccio; Maurizio de Martino
Journal:  BMC Med Genet       Date:  2015-08-23       Impact factor: 2.103

  7 in total

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