Evidence map›Paper›PMID 35490291›Full record

ReviewJournal of inherited metabolic disease2022

Diagnosis and discovery: Insights from the NIH Undiagnosed Diseases Program.

Carolina Montano, Thomas Cassini, Shira G Ziegler, Manfred Boehm, Elena-Raluca Nicoli, Joseph A Mindell, Ariane G Soldatos, Irini Manoli, Lynne Wolfe, Ellen F Macnamara and 5 more

Registry-linked trialOpen access · bronzeAbstract readReview
In one paragraph

Review in Journal of inherited metabolic disease, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT02450851 (Clinical and Genetic Evaluation of Patients With Undiagnosed Disorders Through the Undiagnosed Diseases Network), which is not on this map. Cited by 9 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
9citing papers in PubMed, 1 pooled it
4.7field-weighted citation impact, top 5% of its field
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.

NCT02450851 recruitingnot on this map

Clinical and Genetic Evaluation of Patients With Undiagnosed Disorders Through the Undiagnosed Diseases Network

TypeobservationalSponsorNational Human Genome Research Institute (NHGRI)Ran2015 to 2028Enrolled20,000ConditionsGenetic Disease
3 · Its place in the literature

Who cites it

9 citing papers in PubMed, 1 synthesis or guideline pooled it, 25 citations in OpenAlex.

  1. Pooled it
  2. Article
  3. Review
  4. A genome-wide approach for the discovery of novel repeat expansion disorders in the Undiagnosed Diseases Network cohort.Genetics in medicine : official journal of the American College of Medical Genetics · 2025
    Article
  5. Article
  6. Article
  7. Review
  8. Review
  9. 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

15 authors at 5 institutions in 1 country.

Carolina MontanoMedical Genetics & Genomic Medicine Training Program, National Human Genome Research Institute (NHGRI), NIH, Bethesda, Maryland, USA.ORCID 0000-0002-2772-2025
Thomas CassiniMedical Genetics & Genomic Medicine Training Program, National Human Genome Research Institute (NHGRI), NIH, Bethesda, Maryland, USA.ORCID 0000-0002-3522-2135
Shira G ZieglerDepartments of Pediatrics and Genetic Medicine, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.ORCID 0000-0002-3007-9755
Manfred BoehmLaboratory of Cardiovascular Regenerative Medicine, National Heart, Lung, and Blood Institute (NHLBI), NIH, Bethesda, Maryland, USA.ORCID 0000-0002-2251-3638
Elena-Raluca NicoliGlycosphingolipid and Glycoprotein Disorders Unit, Medical Genetics Branch, National Human Genome Research Institute (NHGRI), NIH, Bethesda, Maryland, USA.ORCID 0000-0002-5545-630X
Joseph A MindellMembrane Transport Biophysics Section, National Institute of Neurological Disorders and Stroke (NINDS), NIH, Bethesda, Maryland, USA.ORCID 0000-0002-6952-8247
Ariane G SoldatosOffice of the Clinical Director, National Institute of Neurological Disorders and Stroke (NINDS), NIH, Bethesda, Maryland, USA.ORCID 0000-0003-3570-5230
Irini ManoliOrganic Acid Research Section, National Human Genome Research Institute (NHGRI), NIH, Bethesda, Maryland, USA.ORCID 0000-0003-1543-2941
Lynne WolfeOffice of the Clinical Director, National Human Genome Research Institute (NHGRI), NIH, Bethesda, Maryland, USA.ORCID 0000-0001-7628-5741
Ellen F MacnamaraNIH Undiagnosed Diseases Program, Common Fund, NIH, Bethesda, Maryland, USA.ORCID 0000-0002-9418-3287
May Christine V MalicdanNIH Undiagnosed Diseases Program, Common Fund, NIH, Bethesda, Maryland, USA.ORCID 0000-0003-1697-6959
David R AdamsOffice of the Clinical Director, National Human Genome Research Institute (NHGRI), NIH, Bethesda, Maryland, USA.
Cynthia J TifftGlycosphingolipid and Glycoprotein Disorders Unit, Medical Genetics Branch, National Human Genome Research Institute (NHGRI), NIH, Bethesda, Maryland, USA.ORCID 0000-0002-3931-1207
Camilo ToroNIH Undiagnosed Diseases Program, Common Fund, NIH, Bethesda, Maryland, USA.
William A GahlOffice of the Clinical Director, National Human Genome Research Institute (NHGRI), NIH, Bethesda, Maryland, USA.ORCID 0000-0002-2494-6752
National Human Genome Research Institute · USOffice of the Director · USNational Institute of Neurological Disorders and Stroke · USJohns Hopkins University · USNational Heart Lung and Blood Institute · US

Funding

Human Biochemical GeneticsZIAHG000215 · NHGRI · NATIONAL HUMAN GENOME RESEARCH INSTITUTE · PI GAHL, WILLIAM · 2009 to 2025
$72.6M
Vascular remodeling in patients with rare genetic disordersZIAHL006077 · NHLBI · NATIONAL HEART, LUNG, AND BLOOD INSTITUTE · PI BOEHM, MANFRED · 2010 to 2025
$22.9M
iPS-technology and patient specific disease modelsZIAHL006079 · NHLBI · NATIONAL HEART, LUNG, AND BLOOD INSTITUTE · PI BOEHM, MANFRED · 2010 to 2025
$14.4M
Chloride fluxes in organellar membranesZIANS003015 · NINDS · NATIONAL INSTITUTE OF NEUROLOGICAL DISORDERS AND STROKE · PI MINDELL, JOSEPH A · 2009 to 2025
$14.2M
Postdoctoral Medical Genetics Training GrantT32GM007471 · NIGMS · JOHNS HOPKINS UNIVERSITY · PI TAO WANG · 1985 to 2026
$8.0M
Pathogenesis informs therapy in the GM1 and GM2 GangliosidosesZIAHG200409 · NHGRI · NATIONAL HUMAN GENOME RESEARCH INSTITUTE · PI TIFFT, CYNTHIA · 2020 to 2025
$4.1M
C BRIGGSAE AND C ELEGANS GENOMIC SEQUENCE COMPARISONF32HG000130 · NHGRI · WASHINGTON UNIVERSITY · PI COUCH, JENNIFER A · 1994 to 1995
–
NHGRI NIH HHS F32 HG000130NIGMS NIH HHS T32 GM007471
6 · The paper itself

Abstract

Living with an undiagnosed medical condition places a tremendous burden on patients, their families, and their healthcare providers. The Undiagnosed Diseases Program (UDP) was established at the National Institutes of Health (NIH) in 2008 with the primary goals of providing a diagnosis for patients with mysterious conditions and advancing medical knowledge about rare and common diseases. The program reviews applications from referring clinicians for cases that are considered undiagnosed despite a thorough evaluation. Those that are accepted receive clinical evaluations involving deep phenotyping and genetic testing that includes exome and genomic sequencing. Selected candidate gene variants are evaluated by collaborators using functional assays. Since its inception, the UDP has received more than 4500 applications and has completed evaluations on nearly 1300 individuals. Here we present six cases that exemplify the discovery of novel disease mechanisms, the importance of deep phenotyping for rare diseases, and how genetic diagnoses have led to appropriate treatment. The creation of the Undiagnosed Diseases Network (UDN) in 2014 has substantially increased the number of patients evaluated and allowed for greater opportunities for data sharing. Expansion to the Undiagnosed Diseases Network International (UDNI) has the possibility to extend this reach even farther. Together, networks of undiagnosed diseases programs are powerful tools to advance our knowledge of pathophysiology, accelerate accurate diagnoses, and improve patient care for patients with rare conditions.

Indexed as

Undiagnosed DiseasesExomeHumansNational Institutes of Health (U.S.)Rare DiseasesUnited StatesUridine DiphosphateUridine DiphosphateamyloidosisCHST14CLCN7exome sequencingKMT2BNT5ESNORD118SNP microarrayUndiagnosed Diseases Programwhole genome sequencing

Identifiers

PMID35490291
PMCPMC12169816
OpenAlexW4225254774

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

Registered trials

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.