Literature DB >> 24462885

Confirmation and further delineation of the 3q26.33-3q27.2 microdeletion syndrome.

Majed Dasouki1, Jennifer Roberts2, Angela Santiago3, Irfan Saadi4, Karine Hovanes5.   

Abstract

Recently, 3 unrelated children with a potentially novel 3q26.33-3q27.2 microdeletion syndrome were reported. We now report a new 9 ½ years old Caucasian boy with a 2 Mb deletion of the same genomic region in combination with Klinefelter syndrome. He presented with facial dysmorphism, developmental delay, Asperger syndrome, thrombocytopenia, recurrent infections and hypogammaglobulinemia. The deletion in our patient improves upon the minimum region of the novel 3q26.33-3q27.2 microdeletion, and provides additional insights into the underlying genetic basis of the observed phenotypes. Consistent with two of three previously described patients, our patient also presents with thrombocytopenia, which we postulate is caused by haploinsufficiency of THPO. In addition, haploinsufficiency of LAMP3, a lymphoid and dendritic cell expressed protein that is implicated in bacterial and viral infections, pulmonary surfactant protein transport and amelogenin degradation, may be a novel cause for the immune deficiency, lung disease and dental abnormalities respectively as seen in these patients.
Copyright © 2014 Elsevier Masson SAS. All rights reserved.

Entities:  

Keywords:  3q26.33–3q27.2 microdeletion syndrome; Array-CGH; Growth retardation; Immunodeficiency; LAMP3; Thrombopoietin/THPO

Mesh:

Year:  2014        PMID: 24462885     DOI: 10.1016/j.ejmg.2013.12.007

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


  7 in total

1.  Monoallelic loss-of-function THPO variants cause heritable thrombocytopenia.

Authors:  Naomi Cornish; M Riyaad Aungraheeta; Lucy FitzGibbon; Kate Burley; Dominic Alibhai; Janine Collins; Daniel Greene; Kate Downes; Sarah K Westbury; Ernest Turro; Andrew D Mumford
Journal:  Blood Adv       Date:  2020-03-10

2.  RhoGTPase Regulators Orchestrate Distinct Stages of Synaptic Development.

Authors:  Samuel Martin-Vilchez; Leanna Whitmore; Hannelore Asmussen; Jessica Zareno; Rick Horwitz; Karen Newell-Litwa
Journal:  PLoS One       Date:  2017-01-23       Impact factor: 3.240

3.  3q27.1 microdeletion causes prenatal and postnatal growth restriction and neurodevelopmental abnormalities.

Authors:  Subit Barua; Elaine M Pereira; Vaidehi Jobanputra; Kwame Anyane-Yeboa; Brynn Levy; Jun Liao
Journal:  Mol Cytogenet       Date:  2022-03-03       Impact factor: 2.009

Review 4.  Inherited platelet disorders: toward DNA-based diagnosis.

Authors:  Claire Lentaigne; Kathleen Freson; Michael A Laffan; Ernest Turro; Willem H Ouwehand
Journal:  Blood       Date:  2016-04-19       Impact factor: 25.476

5.  An 8.4-Mb 3q26.33-3q28 microdeletion in a patient with blepharophimosis-intellectual disability syndrome and a review of the literature.

Authors:  Katrin Õunap; Sander Pajusalu; Olga Zilina; Tiia Reimand; Riina Žordania
Journal:  Clin Case Rep       Date:  2016-07-22

6.  3p22.1p21.31 microdeletion identifies CCK as Asperger syndrome candidate gene and shows the way for therapeutic strategies in chromosome imbalances.

Authors:  Ivan Y Iourov; Svetlana G Vorsanova; Victoria Y Voinova; Yuri B Yurov
Journal:  Mol Cytogenet       Date:  2015-10-31       Impact factor: 2.009

7.  Recessive missense LAMP3 variant associated with defect in lamellar body biogenesis and fatal neonatal interstitial lung disease in dogs.

Authors:  Kati J Dillard; Matthias Ochs; Julia E Niskanen; Meharji Arumilli; Jonas Donner; Kaisa Kyöstilä; Marjo K Hytönen; Marjukka Anttila; Hannes Lohi
Journal:  PLoS Genet       Date:  2020-03-09       Impact factor: 5.917

  7 in total

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