Literature DB >> 36203604

A rapid turnaround gene panel for severe autoinflammation: Genetic results within 48 hours.

Dara McCreary1, Ebun Omoyinmi1,2, Ying Hong1, Barbara Jensen1, Alice Burleigh1,3, Fiona Price-Kuehne1, Kimberly Gilmour4, Despina Eleftheriou1,3,5, Paul Brogan1,5.   

Abstract

There is an important unmet clinical need for fast turnaround next generation sequencing (NGS) to aid genetic diagnosis of patients with acute and sometimes catastrophic inflammatory presentations. This is imperative for patients who require precise and targeted treatment to prevent irreparable organ damage or even death. Acute and severe hyper- inflammation may be caused by primary immunodeficiency (PID) with immune dysregulation, or more typical autoinflammatory diseases in the absence of obvious immunodeficiency. Infectious triggers may be present in either immunodeficiency or autoinflammation. We compiled a list of 25 genes causing monogenetic immunological diseases that are notorious for their acute first presentation with fulminant inflammation and which may be amenable to specific treatment, including hemophagocytic lymphohistiocytosis (HLH); and autoinflammatory diseases that can present with early-onset stroke or other irreversible neurological inflammatory complications. We designed and validated a pipeline that enabled return of clinically actionable results in hours rather than weeks: the Rapid Autoinflammation Panel (RAP). We demonstrated accuracy of this new pipeline, with 100% sensitivity and 100% specificity. Return of results to clinicians was achieved within 48-hours from receiving the patient's blood or saliva sample. This approach demonstrates the potential significant diagnostic impact of NGS in acute medicine to facilitate precision medicine and save "life or limb" in these critical situations.
Copyright © 2022 McCreary, Omoyinmi, Hong, Jensen, Burleigh, Price-Kuehne, Gilmour, Eleftheriou and Brogan.

Entities:  

Keywords:  autoinflammation; genetics; hyper-inflammation; immunodeficiency; turnaround time (TAT)

Mesh:

Year:  2022        PMID: 36203604      PMCID: PMC9531256          DOI: 10.3389/fimmu.2022.998967

Source DB:  PubMed          Journal:  Front Immunol        ISSN: 1664-3224            Impact factor:   8.786


  29 in total

1.  RAPIDOMICS: rapid genome-wide sequencing in a neonatal intensive care unit-successes and challenges.

Authors:  Alison M Elliott; Christèle du Souich; Anna Lehman; Ilaria Guella; Daniel M Evans; Tara Candido; Leah Tooman; Linlea Armstrong; Lorne Clarke; William Gibson; Harinder Gill; Pascal M Lavoie; Suzanne Lewis; Margaret L McKinnon; Sarah M Nikkel; Millan Patel; Alfonso Solimano; Anne Synnes; Joseph Ting; Margot van Allen; Jan Christilaw; Matthew J Farrer; Jan M Friedman; Horacio Osiovich
Journal:  Eur J Pediatr       Date:  2019-06-07       Impact factor: 3.183

2.  Feasibility of Ultra-Rapid Exome Sequencing in Critically Ill Infants and Children With Suspected Monogenic Conditions in the Australian Public Health Care System.

Authors:  Sebastian Lunke; Stefanie Eggers; Meredith Wilson; Chirag Patel; Christopher P Barnett; Jason Pinner; Sarah A Sandaradura; Michael F Buckley; Emma I Krzesinski; Michelle G de Silva; Gemma R Brett; Kirsten Boggs; David Mowat; Edwin P Kirk; Lesley C Adès; Lauren S Akesson; David J Amor; Samantha Ayres; Anne Baxendale; Sarah Borrie; Alessandra Bray; Natasha J Brown; Cheng Yee Chan; Belinda Chong; Corrina Cliffe; Martin B Delatycki; Matthew Edwards; George Elakis; Michael C Fahey; Andrew Fennell; Lindsay Fowles; Lyndon Gallacher; Megan Higgins; Katherine B Howell; Lauren Hunt; Matthew F Hunter; Kristi J Jones; Sarah King; Smitha Kumble; Sarah Lang; Maelle Le Moing; Alan Ma; Dean Phelan; Michael C J Quinn; Anna Richards; Christopher M Richmond; Jessica Riseley; Jonathan Rodgers; Rani Sachdev; Simon Sadedin; Luregn J Schlapbach; Janine Smith; Amanda Springer; Natalie B Tan; Tiong Y Tan; Suzanna L Temple; Christiane Theda; Anand Vasudevan; Susan M White; Alison Yeung; Ying Zhu; Melissa Martyn; Stephanie Best; Tony Roscioli; John Christodoulou; Zornitza Stark
Journal:  JAMA       Date:  2020-06-23       Impact factor: 56.272

3.  Heterozygosity for the common perforin mutation, p.A91V, impairs the cytotoxicity of primary natural killer cells from healthy individuals.

Authors:  Imran G House; Kevin Thia; Amelia J Brennan; Richard Tothill; Alexander Dobrovic; Wei Z Yeh; Richard Saffery; Zac Chatterton; Joseph A Trapani; Ilia Voskoboinik
Journal:  Immunol Cell Biol       Date:  2015-03-17       Impact factor: 5.126

4.  Hereditary Systemic Autoinflammatory Diseases: Therapeutic Stratification.

Authors:  Ovgu Kul Cinar; Amber Putland; Karen Wynne; Despina Eleftheriou; Paul A Brogan
Journal:  Front Pediatr       Date:  2022-04-28       Impact factor: 3.569

5.  Phenotypical heterogeneity in RAG-deficient patients from a highly consanguineous population.

Authors:  S S Meshaal; R E El Hawary; D S Abd Elaziz; A Eldash; R Alkady; S Lotfy; A A Mauracher; L Opitz; J Pachlopnik Schmid; M van der Burg; J Chou; N M Galal; J A Boutros; R Geha; A M Elmarsafy
Journal:  Clin Exp Immunol       Date:  2018-11-04       Impact factor: 4.330

6.  ACMG clinical laboratory standards for next-generation sequencing.

Authors:  Heidi L Rehm; Sherri J Bale; Pinar Bayrak-Toydemir; Jonathan S Berg; Kerry K Brown; Joshua L Deignan; Michael J Friez; Birgit H Funke; Madhuri R Hegde; Elaine Lyon
Journal:  Genet Med       Date:  2013-07-25       Impact factor: 8.822

7.  Targeted gene panel sequencing for the rapid diagnosis of acutely ill infants.

Authors:  Luca Brunelli; Sabrina M Jenkins; James M Gudgeon; Steven B Bleyl; Christine E Miller; Tatiana Tvrdik; Shale A Dames; Betsy Ostrander; Josue A F Daboub; Brandon A Zielinski; Erin K Zinkhan; Hunter R Underhill; Theodore Wilson; Joshua L Bonkowsky; Christian C Yost; Lorenzo D Botto; Justin Jenkins; Theodore J Pysher; Pinar Bayrak-Toydemir; Rong Mao
Journal:  Mol Genet Genomic Med       Date:  2019-06-13       Impact factor: 2.183

8.  A Comparison of Variant Calling Pipelines Using Genome in a Bottle as a Reference.

Authors:  Adam Cornish; Chittibabu Guda
Journal:  Biomed Res Int       Date:  2015-10-11       Impact factor: 3.411

9.  Clinical utility of 24-h rapid trio-exome sequencing for critically ill infants.

Authors:  Huijun Wang; Yanyan Qian; Yulan Lu; Qian Qin; Guoping Lu; Guoqiang Cheng; Ping Zhang; Lin Yang; Bingbing Wu; Wenhao Zhou
Journal:  NPJ Genom Med       Date:  2020-05-05       Impact factor: 8.617

10.  Rapid exome sequencing in PICU patients with new-onset metabolic or neurological disorders.

Authors:  Abigail S Carey; John P Schacht; Christine Umandap; David Fasel; Chunhua Weng; Joshua Cappell; Wendy K Chung; Steven G Kernie
Journal:  Pediatr Res       Date:  2020-03-27       Impact factor: 3.756

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