Evidence map›Paper›PMID 37743782›Full record

ArticleAmerican journal of medical genetics. Part A2024

De novo missense variants in ZBTB47 are associated with developmental delays, hypotonia, seizures, gait abnormalities, and variable movement abnormalities.

Scott K Ward, Alexandrea Wadley, Chun-Hui Anne Tsai, Paul J Benke, Lisa Emrick, Kristen Fisher, Kimberly M Houck, Hongzheng Dai, Undiagnosed Diseases Network, Maria J Guillen Sacoto and 10 more

Erratum issued Registry-linked trialOpen access · bronzeAbstract read
In one paragraph

Article in American journal of medical genetics. Part A, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. 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 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
0.9field-weighted citation impact, top 19% 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

2 citing papers in PubMed, 3 citations in OpenAlex.

  1. Article
  2. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

20 authors at 5 institutions in 2 countries.

Scott K WardDepartment of Molecular and Human Genetics, Baylor College of Medicine (BCM), Houston, Texas, USA.ORCID 0009-0002-6052-1900
Alexandrea WadleyDepartment of Pediatrics, Section of Genetics, University of Oklahoma Health Sciences Center, Oklahoma City, Oklahoma, USA.
Chun-Hui Anne TsaiDepartment of Pediatrics, Section of Genetics, University of Oklahoma Health Sciences Center, Oklahoma City, Oklahoma, USA.
Paul J BenkeJoe DiMaggio Children's Hospital, Hollywood, Florida, USA.
Lisa EmrickDepartment of Pediatrics, Section of Neurology and Developmental Neuroscience, Baylor College of Medicine (BCM), Houston, Texas, USA.
Kristen FisherDepartment of Pediatrics, Section of Neurology and Developmental Neuroscience, Baylor College of Medicine (BCM), Houston, Texas, USA.
Kimberly M HouckDepartment of Pediatrics, Section of Neurology and Developmental Neuroscience, Baylor College of Medicine (BCM), Houston, Texas, USA.
Hongzheng DaiDepartment of Molecular and Human Genetics, Baylor College of Medicine (BCM), Houston, Texas, USA.
Undiagnosed Diseases Network
Maria J Guillen SacotoGeneDx, Gaithersburg, Maryland, USA.
William CraigenDepartment of Molecular and Human Genetics, Baylor College of Medicine (BCM), Houston, Texas, USA.
Kimberly GlaserJoe DiMaggio Children's Hospital, Hollywood, Florida, USA.
David R MurdockDepartment of Molecular and Human Genetics, Baylor College of Medicine (BCM), Houston, Texas, USA.
Luis RohenaDepartment of Pediatrics, Division of Medical Genetics, San Antonio Military Medical Center, San Antonio, Texas, USA.
Karin E M DiderichDepartment of Clinical Genetics, Erasmus MC University Medical Center, Rotterdam, the Netherlands.
Hennie T BruggenwirthDepartment of Clinical Genetics, Erasmus MC University Medical Center, Rotterdam, the Netherlands.
Brendan LeeDepartment of Molecular and Human Genetics, Baylor College of Medicine (BCM), Houston, Texas, USA.
Carlos BacinoDepartment of Molecular and Human Genetics, Baylor College of Medicine (BCM), Houston, Texas, USA.
Lindsay C BurrageDepartment of Molecular and Human Genetics, Baylor College of Medicine (BCM), Houston, Texas, USA.ORCID 0000-0002-5108-8861
Jill A RosenfeldDepartment of Molecular and Human Genetics, Baylor College of Medicine (BCM), Houston, Texas, USA.ORCID 0000-0001-5664-7987
Baylor College of Medicine · USBrooke Army Medical Center · USUniversity of Oklahoma Health Sciences Center · USErasmus MC · NLJoe DiMaggio Children's Hospital · US

Funding

Pilot of New Technologies to Increase the Genomic Diagnosis of Undiagnosed Disease Network (UDN) PatientsU01HG007709 · NHGRI · BAYLOR COLLEGE OF MEDICINE · PI BACINO, CARLOS A., LEE, BRENDAN · 2014 to 2022
$14.3M
Overall: Eunice Kennedy Shriver Intellectual and Developmental Disabilities Research Center at VanderbiltP50HD103537 · NICHD · VANDERBILT UNIVERSITY MEDICAL CENTER · PI Jeffrey L Neul · 2020 to 2026
$10.3M
Preclinical and Clincial OutcomesP50HD103555 · NICHD · BAYLOR COLLEGE OF MEDICINE · PI Sandesh Chakravarthy Sreenath Nagamani, David Loren Nelson · 2020 to 2026
$9.9M
UCLA clinical site for the investigation of undiagnosed disordersU01NS134356 · NINDS · UNIVERSITY OF CALIFORNIA LOS ANGELES · PI MARTINEZ-AGOSTO, JULIAN, NELSON, STANLEY F. · 2023 to 2023
$936k
NCI NIH HHSNHGRI NIH HHS U01 HG007709NICHD NIH HHS P50 HD103537NICHD NIH HHS P50 HD103555NIH HHS U01HG007709NINDS NIH HHS U01 NS134356
6 · The paper itself

Abstract

The collection of known genetic etiologies of neurodevelopmental disorders continues to increase, including several syndromes associated with defects in zinc finger protein transcription factors (ZNFs) that vary in clinical severity from mild learning disabilities and developmental delay to refractory seizures and severe autism spectrum disorder. Here we describe a new neurodevelopmental disorder associated with variants in ZBTB47 (also known as ZNF651), which encodes zinc finger and BTB domain-containing protein 47. Exome sequencing (ES) was performed for five unrelated patients with neurodevelopmental disorders. All five patients are heterozygous for a de novo missense variant in ZBTB47, with p.(Glu680Gly) (c.2039A>G) detected in one patient and p.(Glu477Lys) (c.1429G>A) identified in the other four patients. Both variants impact conserved amino acid residues. Bioinformatic analysis of each variant is consistent with pathogenicity. We present five unrelated patients with de novo missense variants in ZBTB47 and a phenotype characterized by developmental delay with intellectual disability, seizures, hypotonia, gait abnormalities, and variable movement abnormalities. We propose that these variants in ZBTB47 are the basis of a new neurodevelopmental disorder.

Indexed as

Autism Spectrum DisorderDNA-Binding ProteinsIntellectual DisabilityMovement DisordersNeurodevelopmental DisordersTranscription FactorsChildDevelopmental DisabilitiesGaitHumansMuscle HypotoniaPhenotypeSeizuresDNA-Binding ProteinsTranscription FactorsZBTB47 protein, humandevelopmental delayneurodevelopmentalseizureZBTB47zinc fingerZNF651

Identifiers

PMID37743782
PMCPMC11221546
OpenAlexW4387002975

What Socratic holds

Textmetadata
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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 Socratic graph.