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NICUSeq: A Trial to Evaluate the Clinical Utility of Human Whole Genome Sequencing (WGS) Compared to Standard of Care in Acute Care Neonates and Infants

Not Applicable
Completed
Conditions
Rare Diseases
Interventions
Other: clinical whole genome sequencing (cWGS)
Registration Number
NCT03290469
Lead Sponsor
Illumina, Inc.
Brief Summary

Prospective, multi-site, study to evaluate the clinical utility of cWGS in a proband. One group will receive cWGS and a clinical report approximately 15 days after blood samples are received, while the other group will continue to receive standard of care until Day 60. The standard of care group will receive cWGS and a clinical report at Day 60 as part of secondary and tertiary analyses. Both groups will be followed for a total of 90 days.

Detailed Description

This is a prospective, multi-site, randomized study to evaluate the clinical utility of cWGS in each proband. Throughout this study, each proband will receive SOC testing as determined by the site clinical team. Upon enrollment in the study, each proband will be randomly assigned to the 15 day cWGS group or the SOC group. SOC is defined as the management of the proband's care under the same or similar conditions as if the proband was not enrolled in this study. A blood sample from each enrolled proband will be collected and shipped to the Illumina Clinical Services Laboratory ("ICSL"), which is Clinical Laboratory Improvement Amendments (CLIA)-certified and College of American Pathologists (CAP)-accredited. ICSL will conduct cWGS testing with the TruGenome Undiagnosed Disease Test ("TruGenome Test"). The TruGenome Test cWGS results will be provided to the Principal Investigator (PI) or designee who will evaluate each proband test outcome based on the aggregate medical information, informed by the cWGS or SOC results.

Recruitment & Eligibility

Status
COMPLETED
Sex
All
Target Recruitment
355
Inclusion Criteria

Not provided

Exclusion Criteria

Proband Exclusion Criteria

  1. Known non-genetic cause(s) of disease, disorder, or phenotypic defect
  2. The phenotype is fully explained by complications of prematurity
  3. Trisomy 13, 18 or 21 or Turner Syndrome is the likely diagnosis; such a proband will be eligible if a diagnostic karyotype is normal
  4. Blood transfusion within 48 hours (each proband will be re-eligible 48 hours after the most recent transfusion)
  5. The PI decides that the study is not in the best interest of the proband (for example, the neonate or infant is at a high risk of severe morbidity or mortality within the next 7 days and these risks could be mitigated by alternative testing). Subsequent eligibility for enrollment of each proband is at the discretion of the site PI.

Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
15 day cWGS and Standard of Careclinical whole genome sequencing (cWGS)Enrolled cohorts receive the results of the clinical whole genome sequencing (cWGS) after 15 days of the sample receipt while still undergoing standard of care (SOC).
Primary Outcome Measures
NameTimeMethod
A difference in Change of Management between the 15 day cWGS and standard of care groupsDay 60

Change of Management is a binary (yes or no) based on assignments made by the PI or designee at each site using the following domains:

* Condition specific management

* Condition specific supportive interventions

* Palliative care/End of Life Care A change in any of these domains will be considered a change of management.

Secondary Outcome Measures
NameTimeMethod
cWGS satisfaction questionnaire will be given to clinicians and families at the conclusion of the study.90 Days

The questionnaire is a likard scale questionnaire developed by the study team to assess satisfaction levels from the perspective of the clinician and also the parent.

Diagnostic Accuracy90 Days

Diagnostic accuracy (percent positive agreement between test outcome classified by the medical monitor and the site PI or designee)

% diagnoses returned before discharge or death

Average Time to Diagnosis90 Days

Average time (in days) to diagnose between cWGS and SOC based on the comparison of the (a) cWGS results and the (b) current clinical diagnoses

Change in Care Setting from the ICU environment90 Days

Changes in care level setting from the ICU environment will be compared between the 15 day cWGS group and the SOC group.

Diagnostic Yield90 Days

Diagnostic yield (# positive diagnoses/ total # of each proband expressed as a percentage)

Genetic Results Returned90 Days

% diagnoses returned before discharge or death

Costs90 Days

Pre-test costs of hospital care

Time to diagnosis90 Days

Time to diagnosis (in days of life)

The amount of imaging tests ordered as assessed by counting the number of tests per cohort.90 Days

Clinical services utilization includes the number of imaging tests ordered.

Assessment of Clinical Utility by using a questionnaire90 Days

A questionnaire developed by the study team will assess the Clinical Utility of the cWGS test

Trial Locations

Locations (5)

Rady's/Children's Hospital of Orange County

🇺🇸

Orange, California, United States

Children's Hospital of Philadelpia

🇺🇸

Philadelphia, Pennsylvania, United States

Washington University in St. Louis School of Medicine & St. Louis Children's Hospital

🇺🇸

Saint Louis, Missouri, United States

LeBonheur Hospital

🇺🇸

Memphis, Tennessee, United States

University of Nebraska Medical Center & Children's Hospital

🇺🇸

Omaha, Nebraska, United States

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