Literature DB >> 29565416

Exome sequencing in neonates: diagnostic rates, characteristics, and time to diagnosis.

Zöe Powis1, Kelly D Farwell Hagman2, Virginia Speare2, Taylor Cain2, Kirsten Blanco2, Layla S Mowlavi2, Emily M Mayerhofer3, David Tilstra3, Timothy Vedder3, Jesse M Hunter2, Marilyn Tsang2, Lina Gonzalez4, Gerald Vockley4, Sha Tang2.   

Abstract

PURPOSE: Neonatal patients are particularly appropriate for utilization of diagnostic exome sequencing (DES), as many Mendelian diseases are known to present in this period of life but often with complex, heterogeneous features. We attempted to determine the diagnostic rates and features of neonatal patients undergoing DES.
METHODS: The clinical histories and results of 66 neonatal patients undergoing DES were retrospectively reviewed.
RESULTS: Clinical DES identified potentially relevant findings in 25 patients (37.9%). The majority of patients had structural anomalies such as birth defects, dysmorphic features, cardiac, craniofacial, and skeletal defects. The average time for clinical rapid testing was 8 days.
CONCLUSION: Our observations demonstrate the utility of family-based exome sequencing in neonatal patients, including familial cosegregation analysis and comprehensive medical review.

Entities:  

Mesh:

Year:  2018        PMID: 29565416     DOI: 10.1038/gim.2018.11

Source DB:  PubMed          Journal:  Genet Med        ISSN: 1098-3600            Impact factor:   8.822


  15 in total

1.  Children's rare disease cohorts: an integrative research and clinical genomics initiative.

Authors:  Shira Rockowitz; Nicholas LeCompte; Mary Carmack; Andrew Quitadamo; Lily Wang; Meredith Park; Devon Knight; Emma Sexton; Lacey Smith; Beth Sheidley; Michael Field; Ingrid A Holm; Catherine A Brownstein; Pankaj B Agrawal; Susan Kornetsky; Annapurna Poduri; Scott B Snapper; Alan H Beggs; Timothy W Yu; David A Williams; Piotr Sliz
Journal:  NPJ Genom Med       Date:  2020-07-06       Impact factor: 8.617

Review 2.  Genetic testing strategies in the newborn.

Authors:  Jeanne Carroll; Kristen Wigby; Sarah Murray
Journal:  J Perinatol       Date:  2020-05-29       Impact factor: 2.521

3.  Early diagnosis of Pearson syndrome in neonatal intensive care following rapid mitochondrial genome sequencing in tandem with exome sequencing.

Authors:  Lauren S Akesson; Stefanie Eggers; Clare J Love; Belinda Chong; Emma I Krzesinski; Natasha J Brown; Tiong Y Tan; Christopher M Richmond; David R Thorburn; John Christodoulou; Matthew F Hunter; Sebastian Lunke; Zornitza Stark
Journal:  Eur J Hum Genet       Date:  2019-07-29       Impact factor: 4.246

4.  Influence of Genetic Information on Neonatologists' Decisions: A Psychological Experiment.

Authors:  Katharine Press Callahan; John Flibotte; Cara Skraban; Katherine Taylor Wild; Steven Joffe; David Munson; Chris Feudtner
Journal:  Pediatrics       Date:  2022-03-01       Impact factor: 7.124

Review 5.  [Application of RNA sequencing in clinical diagnosis of Mendelian disease].

Authors:  Hui Xiao; Wen-Hao Zhou
Journal:  Zhongguo Dang Dai Er Ke Za Zhi       Date:  2020-10

6.  Exome and genome sequencing for pediatric patients with congenital anomalies or intellectual disability: an evidence-based clinical guideline of the American College of Medical Genetics and Genomics (ACMG).

Authors:  Kandamurugu Manickam; Monica R McClain; Laurie A Demmer; Sawona Biswas; Hutton M Kearney; Jennifer Malinowski; Lauren J Massingham; Danny Miller; Timothy W Yu; Fuki M Hisama
Journal:  Genet Med       Date:  2021-07-01       Impact factor: 8.822

Review 7.  Genetic testing for unexplained perinatal disorders.

Authors:  Thomas Hays; Ronald J Wapner
Journal:  Curr Opin Pediatr       Date:  2021-04-01       Impact factor: 2.856

Review 8.  Cancer Genetic Counseling-Current Practice and Future Challenges.

Authors:  Jaclyn Schienda; Jill Stopfer
Journal:  Cold Spring Harb Perspect Med       Date:  2020-06-01       Impact factor: 5.159

Review 9.  Diagnosing rare diseases after the exome.

Authors:  Laure Frésard; Stephen B Montgomery
Journal:  Cold Spring Harb Mol Case Stud       Date:  2018-12-17

10.  Children's rare disease cohorts: an integrative research and clinical genomics initiative.

Authors:  Shira Rockowitz; Nicholas LeCompte; Mary Carmack; Andrew Quitadamo; Lily Wang; Meredith Park; Devon Knight; Emma Sexton; Lacey Smith; Beth Sheidley; Michael Field; Ingrid A Holm; Catherine A Brownstein; Pankaj B Agrawal; Susan Kornetsky; Annapurna Poduri; Scott B Snapper; Alan H Beggs; Timothy W Yu; David A Williams; Piotr Sliz
Journal:  NPJ Genom Med       Date:  2020-07-06       Impact factor: 8.617

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