Literature DB >> 24311450

Inherited dup(17)(p11.2p11.2): expanding the phenotype of the Potocki-Lupski syndrome.

Pilar L Magoulas1, Pengfei Liu, Violet Gelowani, Claudia Soler-Alfonso, Emma C Kivuva, James R Lupski, Lorraine Potocki.   

Abstract

Potocki-Lupski syndrome (PTLS, OMIM: 610883) is a microduplication syndrome characterized by infantile hypotonia, failure to thrive, cardiovascular malformations, developmental delay, intellectual disability, and behavior abnormalities, the latter of which can include autism spectrum disorder. The majority of individuals with PTLS harbor a de novo microduplication of chromosome 17p11.2 reciprocal to the common recurrent 3.6 Mb microdeletion in the Smith-Magenis syndrome critical region. Here, we report on the transmission of the PTLS duplication across two generations in two separate families. Individuals in these families presented initially with developmental delay, behavior problems, and intellectual disability. We provide a detailed review of the clinical and developmental phenotype of inherited PTLS in both families. This represents the second report (second and third families) of PTLS in a parent-child pair and exemplifies the under-diagnosis of this and likely other genetic conditions in adults with intellectual disability and/or psychiatric disorders.
© 2013 Wiley Periodicals, Inc.

Entities:  

Keywords:  PTLS; Potocki-Lupski syndrome; RAI1; dosage sensitivity; microduplication syndrome

Mesh:

Year:  2013        PMID: 24311450     DOI: 10.1002/ajmg.a.36287

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  9 in total

Review 1.  Neurodevelopmental Disorders Associated with Abnormal Gene Dosage: Smith-Magenis and Potocki-Lupski Syndromes.

Authors:  Juanita Neira-Fresneda; Lorraine Potocki
Journal:  J Pediatr Genet       Date:  2015-09-28

2.  A New Patient with Potocki-Lupski Syndrome: A Literature Review.

Authors:  Andrea Domenico Praticò; Raffaele Falsaperla; Renata Rizzo; Martino Ruggieri; Alberto Verrotti; Piero Pavone
Journal:  J Pediatr Genet       Date:  2017-07-27

3.  Unusual de novo Partial Trisomy 17p12p11.2 due to Unbalanced Insertion into 5p13.1 in a Severely Affected Boy.

Authors:  Luis Alberto Mendez-Rosado; Araceli Lantigua; Juan Galarza; Ahmed B Hamid Al-Rikabi; Monika Ziegler; Thomas Liehr
Journal:  J Pediatr Genet       Date:  2017-03-07

4.  RAI1 Overexpression Promotes Altered Circadian Gene Expression and Dyssomnia in Potocki-Lupski Syndrome.

Authors:  Sureni V Mullegama; Joseph T Alaimo; Michael D Fountain; Brooke Burns; Amanda Hebert Balog; Li Chen; Sarah H Elsea
Journal:  J Pediatr Genet       Date:  2017-03-07

5.  Nonrecurrent 17p11.2p12 Rearrangement Events that Result in Two Concomitant Genomic Disorders: The PMP22-RAI1 Contiguous Gene Duplication Syndrome.

Authors:  Bo Yuan; Tamar Harel; Shen Gu; Pengfei Liu; Lydie Burglen; Sandra Chantot-Bastaraud; Violet Gelowani; Christine R Beck; Claudia M B Carvalho; Sau Wai Cheung; Andrew Coe; Valérie Malan; Arnold Munnich; Pilar L Magoulas; Lorraine Potocki; James R Lupski
Journal:  Am J Hum Genet       Date:  2015-11-05       Impact factor: 11.025

Review 6.  Trans-acting epigenetic effects of chromosomal aneuploidies: lessons from Down syndrome and mouse models.

Authors:  Catherine Do; Zhuo Xing; Y Eugene Yu; Benjamin Tycko
Journal:  Epigenomics       Date:  2016-12-02       Impact factor: 4.778

7.  Prenatal Diagnosis of 17p11.2 Copy Number Abnormalities Associated With Smith-Magenis and Potocki-Lupski Syndromes in Fetuses.

Authors:  Meiying Cai; Xianguo Fu; Liangpu Xu; Na Lin; Hailong Huang
Journal:  Front Genet       Date:  2021-12-21       Impact factor: 4.599

8.  Anesthesia in a Patient with Potocki-Lupski Syndrome.

Authors:  Songhyun Kim; Yunhee Lim; In-Jung Jun; Byunghoon Yoo; Kye-Min Kim
Journal:  Case Rep Anesthesiol       Date:  2021-12-04

9.  Copy number loss upstream of RAI1 uncovers gene expression regulatory region that may impact Potocki-Lupski syndrome diagnosis.

Authors:  Joseph T Alaimo; Sureni V Mullegama; Mary Ann Thomas; Sarah H Elsea
Journal:  Mol Cytogenet       Date:  2015-10-05       Impact factor: 2.009

  9 in total

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