Literature DB >> 22548977

Molecular cytogenetic characterization of an interstitial deletion of chromosome 21 (21q22.13q22.3) in a patient with dysmorphic features, intellectual disability and severe generalized epilepsy.

Angelo Valetto1, Alessandro Orsini, Veronica Bertini, Benedetta Toschi, Alice Bonuccelli, Francesca Simi, Irene Sammartino, Grazia Taddeucci, Paolo Simi, Giuseppe Saggese.   

Abstract

We report on a de novo interstitial deletion of chromosome 21q in a patient presenting with characteristic facial features, intellectual disability, and epilepsy. The deletion extent was about 4.9 Mb from position 37713441 bp (21q22.13) to position 42665162 bp (21q22.3) (NCBI36/hg18 map). Patients with partial monosomy 21 are quite rare; this anomaly has been associated with a wide spectrum of clinical signs, ranging from very mild to quite severe phenotypes. This variability results from variability in the deleted regions, thus accurate molecular definition of the chromosomal breakpoints is necessary to make better genotype-phenotype correlations. We compared our patient's phenotype with the few other patients reported in the literature and found to have similar deletion when analyzed by array CGH. The minimal overlapping region contains only two genes, DYRK1A and KCNJ6, which may play a major role in these patients' phenotype.
Copyright © 2012 Elsevier Masson SAS. All rights reserved.

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Year:  2012        PMID: 22548977     DOI: 10.1016/j.ejmg.2012.03.011

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


  19 in total

1.  Ten new cases further delineate the syndromic intellectual disability phenotype caused by mutations in DYRK1A.

Authors:  Lucas M Bronicki; Claire Redin; Severine Drunat; Amélie Piton; Michael Lyons; Sandrine Passemard; Clarisse Baumann; Laurence Faivre; Julien Thevenon; Jean-Baptiste Rivière; Bertrand Isidor; Grace Gan; Christine Francannet; Marjolaine Willems; Murat Gunel; Julie R Jones; Joseph G Gleeson; Jean-Louis Mandel; Roger E Stevenson; Michael J Friez; Arthur S Aylsworth
Journal:  Eur J Hum Genet       Date:  2015-04-29       Impact factor: 4.246

2.  DYRK1A haploinsufficiency causes a new recognizable syndrome with microcephaly, intellectual disability, speech impairment, and distinct facies.

Authors:  Jianling Ji; Hane Lee; Bob Argiropoulos; Naghmeh Dorrani; John Mann; Julian A Martinez-Agosto; Natalia Gomez-Ospina; Natalie Gallant; Jonathan A Bernstein; Louanne Hudgins; Leah Slattery; Bertrand Isidor; Cédric Le Caignec; Albert David; Ewa Obersztyn; Barbara Wiśniowiecka-Kowalnik; Michelle Fox; Joshua L Deignan; Eric Vilain; Emily Hendricks; Margaret Horton Harr; Sarah E Noon; Jessi R Jackson; Alisha Wilkens; Ghayda Mirzaa; Noriko Salamon; Jeff Abramson; Elaine H Zackai; Ian Krantz; A Micheil Innes; Stanley F Nelson; Wayne W Grody; Fabiola Quintero-Rivera
Journal:  Eur J Hum Genet       Date:  2015-05-06       Impact factor: 4.246

3.  Development and Validation of a Deep Learning Model to Screen for Trisomy 21 During the First Trimester From Nuchal Ultrasonographic Images.

Authors:  Liwen Zhang; Di Dong; Yongqing Sun; Chaoen Hu; Congxin Sun; Qingqing Wu; Jie Tian
Journal:  JAMA Netw Open       Date:  2022-06-01

4.  Social motivation a relative strength in DYRK1A syndrome on a background of significant speech and language impairments.

Authors:  Lottie D Morison; Ruth O Braden; David J Amor; Amanda Brignell; Bregje W M van Bon; Angela T Morgan
Journal:  Eur J Hum Genet       Date:  2022-04-18       Impact factor: 5.351

5.  G Protein-Gated K+ Channel Ablation in Forebrain Pyramidal Neurons Selectively Impairs Fear Learning.

Authors:  Nicole C Victoria; Ezequiel Marron Fernandez de Velasco; Olga Ostrovskaya; Stefania Metzger; Zhilian Xia; Lydia Kotecki; Michael A Benneyworth; Anastasia N Zink; Kirill A Martemyanov; Kevin Wickman
Journal:  Biol Psychiatry       Date:  2015-11-10       Impact factor: 13.382

6.  Ocular Phenotype Associated with DYRK1A Variants.

Authors:  Cécile Méjécase; Christopher M Way; Nicholas Owen; Mariya Moosajee
Journal:  Genes (Basel)       Date:  2021-02-05       Impact factor: 4.096

7.  Performance of chromosomal microarray for patients with intellectual disabilities/developmental delay, autism, and multiple congenital anomalies in a Chinese cohort.

Authors:  Wilson Wai Sing Chong; Ivan Fai Man Lo; Stephen Tak Sum Lam; Chi Chiu Wang; Ho Ming Luk; Tak Yeung Leung; Kwong Wai Choy
Journal:  Mol Cytogenet       Date:  2014-05-23       Impact factor: 2.009

Review 8.  Case report of novel DYRK1A mutations in 2 individuals with syndromic intellectual disability and a review of the literature.

Authors:  Stephanie M Luco; Daniela Pohl; Erick Sell; Justin D Wagner; David A Dyment; Hussein Daoud
Journal:  BMC Med Genet       Date:  2016-02-27       Impact factor: 2.103

9.  Suppression of pyramidal neuron G protein-gated inwardly rectifying K+ channel signaling impairs prelimbic cortical function and underlies stress-induced deficits in cognitive flexibility in male, but not female, mice.

Authors:  Eden M Anderson; Steven Loke; Benjamin Wrucke; Annabel Engelhardt; Skyler Demis; Kevin O'Reilly; Evan Hess; Kevin Wickman; Matthew C Hearing
Journal:  Neuropsychopharmacology       Date:  2021-06-23       Impact factor: 7.853

10.  Double deletion of a chromosome 21 inserted in a chromosome 22 in an azoospermic patient.

Authors:  Valentine Marquet; Dominique Bourgeois; Philippe De Mas; Laurence Bouneau; Adeline Vigouroux-Castera; Romain Molignier; Patrick Calvas
Journal:  Clin Case Rep       Date:  2015-08-20
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