Literature DB >> 35707596

A New Case of Rare Microdeletion 10q22.3q23 along with Mosaic Klinefelter Syndrome Associated with Facial Dysmorphic Finding, Atrial Ventricular Septal Defect, and Motor Retardation.

Firdevs Dincsoy Bir1, Fatma Silan2, Jelena Velickovic3, Mehmet Berkay Akcan2, Ozturk Ozdemir2.   

Abstract

The chromosome 10q22.3q23.2 deletion syndrome is characterized by craniofacial dysmorphic features, developmental delay, congenital heart defect, and hand/foot abnormalities. In this study, we report a patient carrying a microdeletion of 7.5 Mb at 10q22.3q23.2 and in addition a mosaicism mos 47,XXY[47]/46,XY[23]. This male patient was 3 years and 3 months years old at the time of genetic evaluation. Atrial ventricular septal defect (AVSD), mild hypotonia, torticollis, and left-sided club foot were noticed after birth. The boy had surgical correction of the AVSD and the club foot. His dysmorphic features were frontal bossing, overfolded ear helix, hypertelorism, epicanthal folds, broad base of nose, flat nasal bridge, full cheeks, thick lips, micrognathia, and joint hyperextensibility. His speech/language development was delayed. Klinefelter syndrome is one of the most common congenital chromosomal abnormalities, but usually it is detected in puberty or in adulthood when reproductive failure occurs. Deletions in the 10q22.3q23.2 region are rare, and previously only a few numbers of cases were described with this microdeletion, but none of them together with Klinefelter syndrome and it could be associated with our case clinical features. The new case described will improve understanding the phenotype associated with 10q22.3q23.2 microdeletions. By presenting this case, we aimed to improve the understanding of the phenotype caused by the rare 10q22.3q23.2 deletion and to show the rare coexistence of this deletion with Klinefelter syndrome.
Copyright © 2022 by S. Karger AG, Basel.

Entities:  

Keywords:  10q22.3q23.2 microdeletion; Atrial ventricular septal defect; Klinefelter syndrome; Language delay; Mosaicism

Year:  2022        PMID: 35707596      PMCID: PMC9149477          DOI: 10.1159/000519965

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  24 in total

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Journal:  Pediatrics       Date:  1988-06       Impact factor: 7.124

2.  The phenotype of recurrent 10q22q23 deletions and duplications.

Authors:  Bregje W M van Bon; Jorune Balciuniene; Gary Fruhman; Sandesh Chakravarthy Sreenath Nagamani; Diane L Broome; Elizabeth Cameron; Danielle Martinet; Eliane Roulet; Sebastien Jacquemont; Jacques S Beckmann; Mira Irons; Lorraine Potocki; Brendan Lee; Sau Wai Cheung; Ankita Patel; Melissa Bellini; Angelo Selicorni; Roberto Ciccone; Margherita Silengo; Annalisa Vetro; Nine V Knoers; Nicole de Leeuw; Rolph Pfundt; Barry Wolf; Petr Jira; Swaroop Aradhya; Pawel Stankiewicz; Han G Brunner; Orsetta Zuffardi; Scott B Selleck; James R Lupski; Bert B A de Vries
Journal:  Eur J Hum Genet       Date:  2011-01-19       Impact factor: 4.246

3.  A case with 47,XXY,del(15)(q11;q13) karyotype associated with Prader-Willi phenotype.

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Journal:  Horm Res       Date:  1997

4.  The XXY (Klinefelter's) syndrome in childhood: detection and treatment.

Authors:  P D Caldwell; D W Smith
Journal:  J Pediatr       Date:  1972-02       Impact factor: 4.406

5.  Incontinentia pigmenti, an x-linked dominant disorder, in a 2-year-old boy with Klinefelter syndrome.

Authors:  Abhilasha Williams; Laxmisha Chandrashekar; Vivi M Srivastava; Meera Thomas; Saban Horo; Renu George
Journal:  Indian J Pathol Microbiol       Date:  2017 Jul-Sep       Impact factor: 0.740

Review 6.  Chromosomal variants in klinefelter syndrome.

Authors:  A Frühmesser; D Kotzot
Journal:  Sex Dev       Date:  2011-04-29       Impact factor: 1.824

7.  Down-Klinefelter syndrome (48,XXY,+21) in a neonate associated with congenital heart disease.

Authors:  M A Rodrigues; L F Morgade; L F A Dias; R V Moreira; P D Maia; A F H Sales; P D Ribeiro
Journal:  Genet Mol Res       Date:  2017-09-27

8.  Exome sequencing identifies rare variants in multiple genes in atrioventricular septal defect.

Authors:  Lisa C A D'Alessandro; Saeed Al Turki; Ashok Kumar Manickaraj; Dorin Manase; Barbara J M Mulder; Lynn Bergin; Herschel C Rosenberg; Tapas Mondal; Elaine Gordon; Jane Lougheed; John Smythe; Koen Devriendt; Shoumo Bhattacharya; Hugh Watkins; Jamie Bentham; Sarah Bowdin; Matthew E Hurles; Seema Mital
Journal:  Genet Med       Date:  2015-05-21       Impact factor: 8.822

Review 9.  Update On The Clinical Perspectives And Care Of The Child With 47,XXY (Klinefelter Syndrome).

Authors:  Carole A Samango-Sprouse; Debra R Counts; Selena L Tran; Patricia C Lasutschinkow; Grace F Porter; Andrea L Gropman
Journal:  Appl Clin Genet       Date:  2019-10-23

10.  The distributions of protein coding genes within chromatin domains in relation to human disease.

Authors:  Enrique M Muro; Jonas Ibn-Salem; Miguel A Andrade-Navarro
Journal:  Epigenetics Chromatin       Date:  2019-12-05       Impact factor: 4.954

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