Literature DB >> 32959227

Clinical Utility of Next-Generation Sequencing for Developmental Disorders in the Rehabilitation Department: Experiences from a Single Chinese Center.

Yun Liu1, Xiaomei Liu1, Dongdong Qin2, Yiming Zhao3, Xuanlan Cao1, Xiaoli Deng1, Yu Cheng1, Fuping Liu1, Fang Yang4, Tiesong Zhang5, Xiu-An Yang6.   

Abstract

This study investigated the clinical and genetic characteristics of developmental disorders (DDs) in children attending a rehabilitation department. A total of 94 children with suspected rare and undiagnosed DDs were included in this study. All patients were subjected to next-generation sequencing by means of proband single whole-exome sequencing (Pro-WES) or trio whole-exome sequencing (Trio-WES). To investigate the copy number variations (CNVs), 63 patients were subjected to the trio strategy, and 17 cases were subjected to the proband single strategy. The patients developed early and suffered from severe symptoms. WES reached a high diagnostic rate (48.7%, 46/94), and de novo (48.3%, 28/58) was the main pathogenic form. Most identified single-nucleotide variations (SNVs)/small insertions and deletions (indels) were found only in one patient. The number of uncertain significant locus in the patients taking Trio-WES was significantly lower than that in patients taking Pro-WES (2.1% vs 2.8%). Compared with hereditary mutations passed from parents, pathogenicity was more obvious in de novo mutations. The diagnostic rate of WES accompanied by CNVseq (57.5%, 46/80) was significantly higher (p = 0.016) than WES alone. Next-generation sequencing exhibited a satisfactory diagnostic rate for DDs patients in the rehabilitation department. Compared with the proband-only model, the family trio strategy should be employed more frequently because it can reduce the number of uncertain significant sites and help to identify de novo pathogenic mutations.

Entities:  

Keywords:  Copy number variation sequencing; Developmental disorders; Next-generation sequencing; Rehabilitation department; Whole-genome sequencing

Year:  2020        PMID: 32959227     DOI: 10.1007/s12031-020-01707-4

Source DB:  PubMed          Journal:  J Mol Neurosci        ISSN: 0895-8696            Impact factor:   3.444


  22 in total

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Authors:  Qingguo Jiao; Haiming Sun; Haoya Zhang; Ran Wang; Suting Li; Dan Sun; Xiu-An Yang; Yan Jin
Journal:  Clin Genet       Date:  2019-04-22       Impact factor: 4.438

3.  Diagnostic Yields of Trio-WES Accompanied by CNVseq for Rare Neurodevelopmental Disorders.

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Journal:  Front Genet       Date:  2019-05-24       Impact factor: 4.599

4.  Efficacy and safety of everolimus for subependymal giant cell astrocytomas associated with tuberous sclerosis complex (EXIST-1): a multicentre, randomised, placebo-controlled phase 3 trial.

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Review 5.  Exome sequence read depth methods for identifying copy number changes.

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7.  A copy number variation morbidity map of developmental delay.

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Journal:  Nat Genet       Date:  2011-08-14       Impact factor: 38.330

Review 8.  Psychomotor retardation in depression: a systematic review of diagnostic, pathophysiologic, and therapeutic implications.

Authors:  Djamila Bennabi; Pierre Vandel; Charalambos Papaxanthis; Thierry Pozzo; Emmanuel Haffen
Journal:  Biomed Res Int       Date:  2013-10-30       Impact factor: 3.411

9.  Genomic diagnosis for children with intellectual disability and/or developmental delay.

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Journal:  Genome Med       Date:  2017-05-30       Impact factor: 11.117

10.  Homozygous and hemizygous CNV detection from exome sequencing data in a Mendelian disease cohort.

Authors:  Tomasz Gambin; Zeynep C Akdemir; Bo Yuan; Shen Gu; Theodore Chiang; Claudia M B Carvalho; Chad Shaw; Shalini Jhangiani; Philip M Boone; Mohammad K Eldomery; Ender Karaca; Yavuz Bayram; Asbjørg Stray-Pedersen; Donna Muzny; Wu-Lin Charng; Vahid Bahrambeigi; John W Belmont; Eric Boerwinkle; Arthur L Beaudet; Richard A Gibbs; James R Lupski
Journal:  Nucleic Acids Res       Date:  2017-02-28       Impact factor: 16.971

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