Literature DB >> 28413209

Duplication 17p11.2 (Potocki-Lupski Syndrome) in a child with developmental delay.

S Shuib1, N N Saaid, Z Zakaria, J Ismail, Z Abdul Latiff.   

Abstract

Potocki-Lupski syndrome (PTLS), also known as duplication 17p11.2 syndrome, trisomy 17p11.2 or dup(17)(p11.2p11.2) syndrome, is a developmental disorder and a rare contiguous gene syndrome affecting 1 in 20,000 live births. Among the key features of such patients are autism spectrum disorder, learning disabilities, developmental delay, attention-deficit disorder, infantile hypotonia and cardiovascular abnormalities. Previous studies using microarray identified variations in the size and extent of the duplicated region of chromosome 17p11.2. However, there are a few genes which are considered as candidates for PTLS which include RAI1, SREBF1, DRG2, LLGL1, SHMT1 and ZFP179. In this report, we investigated a case of a 3-year-old girl who has developmental delay. Her chromosome analysis showed a normal karyotype (46,XX). Analysis using array CGH (4X44 K, Agilent USA) identified an ~4.2 Mb de novo duplication in chromosome 17p11.2. The result was confirmed by fluorescence in situ hybridization (FISH) using probes in the critical PTLS region. This report demonstrates the importance of microarray and FISH in the diagnosis of PTLS.

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Year:  2017        PMID: 28413209

Source DB:  PubMed          Journal:  Malays J Pathol        ISSN: 0126-8635            Impact factor:   0.656


  4 in total

1.  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

2.  A unique Smith-Magenis patient with a de novo intragenic deletion on the maternally inherited overexpressed RAI1 allele.

Authors:  Alessandra Sironi; Ilaria Bestetti; Maura Masciadri; Francesca Tumiatti; Milena Crippa; Chiara Pantaleoni; Silvia Russo; Stefano D'Arrigo; Donatella Milani; Lidia Larizza; Palma Finelli
Journal:  Eur J Hum Genet       Date:  2022-07-11       Impact factor: 5.351

Review 3.  Developmentally regulated GTPases: structure, function and roles in disease.

Authors:  Christian A E Westrip; Qinqin Zhuang; Charlotte Hall; Charlotte D Eaton; Mathew L Coleman
Journal:  Cell Mol Life Sci       Date:  2021-10-19       Impact factor: 9.261

4.  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

  4 in total

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