Evidence map›Paper›PMID 33631351›Full record

ArticleThe Journal of molecular diagnostics : JMD2021

Harmonizing the Collection of Clinical Data on Genetic Testing Requisition Forms to Enhance Variant Interpretation in Hypertrophic Cardiomyopathy (HCM): A Study from the ClinGen Cardiomyopathy Variant Curation Expert Panel.

Ana Morales, Alexander Ing, Christian Antolik, Christina Austin-Tse, Linnea M Baudhuin, Lucas Bronicki, Allison Cirino, Megan H Hawley, Michael Fietz, John Garcia and 22 more

Abstract read
In one paragraph

Article in The Journal of molecular diagnostics : JMD, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

32 authors.

Ana MoralesDivision of Human Genetics, The Ohio State University, Columbus, Ohio; Invitae Corp., San Francisco, California. Electronic address: ana.morales@invitae.com.
Alexander IngLaboratory for Molecular Medicine, Partners HealthCare, Boston, Massachusetts.
Christian AntolikDepartment of Clinical Diagnostics, Ambry Genetics, Aliso Viejo, California.
Christina Austin-TseLaboratory for Molecular Medicine, Partners HealthCare, Boston, Massachusetts; Department of Pathology, Massachusetts General Hospital, Boston, Massachusetts; Harvard Medical School, Boston, Massachusetts.
Linnea M BaudhuinDepartment of Laboratory Medicine and Pathology, Mayo Clinic, Rochester, Minnesota.
Lucas BronickiDepartment of Genetics, CHEO, Ottawa, Ontario, Canada; Department of Pathology and Laboratory Medicine, University of Ottawa, Ottawa, Ontario, Canada.
Allison CirinoCardiovascular Division, Brigham and Women's Hospital, Boston, Massachusetts.
Megan H HawleyLaboratory for Molecular Medicine, Partners HealthCare, Boston, Massachusetts.
Michael FietzPathWest Laboratory Medicine, Sir Charles Gairdner Hospital, Nedlands, Western Australia, Australia; Illumina Inc., Scoresby, Victoria, Australia.
John GarciaInvitae Corp., San Francisco, California.
Carolyn HoCardiovascular Division, Departments of Medicine and Radiology, Brigham and Women's Hospital, Boston, Massachusetts.
Jodie InglesAgnes Ginges Centre for Molecular Cardiology at Centenary Institute, The University of Sydney, Sydney, New South Wales, Australia.
Olga JarinovaDepartment of Genetics, CHEO, Ottawa, Ontario, Canada; Department of Pathology and Laboratory Medicine, University of Ottawa, Ottawa, Ontario, Canada.
Tami JohnstonDepartment of Clinical Diagnostics, Ambry Genetics, Aliso Viejo, California.
Melissa A KellyGeisinger, Danville, Pennsylvania.
C Lisa KurtzDepartment of Genetics, University of North Carolina, Chapel Hill, North Carolina.
Matt LeboLaboratory for Molecular Medicine, Partners HealthCare, Boston, Massachusetts; Department of Clinical Diagnostics, Ambry Genetics, Aliso Viejo, California; Department of Pathology, Massachusetts General Hospital, Boston, Massachusetts.
Daniela MacayaGeneDx, Inc, Gaithersburg, Maryland.
Lisa MahantaLaboratory for Molecular Medicine, Partners HealthCare, Boston, Massachusetts.
Joseph MaleszewskiHarvard Medical School, Boston, Massachusetts; Cardiovascular Medicine, Mayo Clinic, Rochester, Minnesota.
Arjun K ManraiComputational Health Informatics Program, Boston Children's Hospital and Harvard Medical School, Boston, Massachusetts.
Mitzi MurrayGeneDx, Inc, Gaithersburg, Maryland.
Gabriele RichardGeneDx, Inc, Gaithersburg, Maryland.
Chris SemsarianCardiovascular Division, Departments of Medicine and Radiology, Brigham and Women's Hospital, Boston, Massachusetts.
Kate L ThomsonOxford Medical Genetics Laboratories, Churchill Hospital, Oxford, United Kingdom.
Tom WinderInvitae Corp., San Francisco, California.
James S WareNational Heart and Lung Institute, Imperial College London, London, United Kingdom; Cardiovascular Research Centre, Royal Brompton and Harefield NHS Foundation Trust, London, London, United Kingdom; MRC London Institute of Medical Sciences, Imperial College London, London, United Kingdom.
Ray E HershbergerDivision of Human Genetics, The Ohio State University, Columbus, Ohio; Division of Cardiovascular Medicine, The Ohio State University, Columbus, Ohio.
Birgit H FunkeLaboratory for Molecular Medicine, Partners HealthCare, Boston, Massachusetts; Department of Clinical Diagnostics, Ambry Genetics, Aliso Viejo, California; Department of Pathology, Massachusetts General Hospital, Boston, Massachusetts.
Matteo VattaInvitae Corp., San Francisco, California; Departments of Medical and Molecular Genetics, Indiana University School of Medicine, Indianapolis, Indiana.
ClinGen Cardiovascular Clinical Domain Working Group
Cardiomyopathy Variant Curation Expert Panel

Funding

Resource ProjectU41HG006834 · NHGRI · BRIGHAM AND WOMEN'S HOSPITAL · PI REHM, HEIDI L · 2013 to 2020
$26.9M
The Future of PharmGKB Funding Under NHGRIU41HG009649 · NHGRI · STANFORD UNIVERSITY · PI BUSTAMANTE, CARLOS DANIEL · 2017 to 2020
$13.4M
The Clinical Genome Resource - Expert Curation and EHR IntegrationU41HG009650 · NHGRI · UNIV OF NORTH CAROLINA CHAPEL HILL · PI BERG, JONATHAN S · 2017 to 2020
$13.3M
Primary Mitochondrial Disease Expert Curation PanelU24HD093483 · NICHD · CHILDREN'S HOSP OF PHILADELPHIA · PI FALK, MARNI J, GAI, XIAOWU · 2017 to 2023
$2.2M
Expert Panel: Genetics of Malformations of the Central Nervous SystemU24HD093487 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI PODURI, ANNAPURNA, YU, TIMOTHY WEI-WEN · 2017 to 2019
$988k
Genomic Clinical Variant Expert Curation Panel for Monogenic Diabetes: A Gateway to Precision Medicine in Diabetes MellitusU24HD093486 · NICHD · UNIVERSITY OF MARYLAND BALTIMORE · PI POLLIN, TONI I. · 2017 to 2019
$816k
Precision Cardiovascular Medicine for Multi-Ethnic PopulationsK01HL138259 · NHLBI · HARVARD MEDICAL SCHOOL · PI MANRAI, ARJUN KUMAR · 2018 to 2022
$801k
Medical Research Council MC_UP_1102/20NHGRI NIH HHS U41 HG006834NHGRI NIH HHS U41 HG009649NHGRI NIH HHS U41 HG009650NHLBI NIH HHS K01 HL138259NICHD NIH HHS U24 HD093483NICHD NIH HHS U24 HD093486NICHD NIH HHS U24 HD093487
6 · The paper itself

Abstract

Diagnostic laboratories gather phenotypic data through requisition forms, but there is no consensus as to which data are essential for variant interpretation. The ClinGen Cardiomyopathy Variant Curation Expert Panel defined a phenotypic data set for hypertrophic cardiomyopathy (HCM) variant interpretation, with the goal of standardizing requisition forms. Phenotypic data elements listed on requisition forms from nine leading cardiomyopathy testing laboratories were compiled to assess divergence in data collection. A pilot of 50 HCM cases was implemented to determine the feasibility of harmonizing data collection. Laboratory directors were surveyed to gauge potential for adoption of a minimal data set. Wide divergence was observed in the phenotypic data fields in requisition forms. The 50-case pilot showed that although demographics and assertion of a clinical diagnosis of HCM had 86% to 98% completion, specific phenotypic features, such as degree of left ventricular hypertrophy, ejection fraction, and suspected syndromic disease, were completed only 24% to 44% of the time. Nine data elements were deemed essential for variant classification by the expert panel. Participating laboratories unanimously expressed a willingness to adopt these data elements in their requisition forms. This study demonstrates the value of comparing and sharing best practices through an expert group, such as the ClinGen Program, to enhance variant interpretation, providing a foundation for leveraging cumulative case-level data in public databases and ultimately improving patient care.

Indexed as

Databases, GeneticGenetic VariationGenome, HumanAdultCardiomyopathy, HypertrophicFemaleGenetic TestingGenomicsHumansMaleMiddle AgedPhenotypeRetrospective Studies

Identifiers

PMID33631351
PMCPMC8188618

What OpenQuestion holds

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.