Evidence map›Paper›PMID 28777491›Full record

ArticleAmerican journal of medical genetics. Part A2017

Identification of STAC3 variants in non-Native American families with overlapping features of Carey-Fineman-Ziter syndrome and Moebius syndrome.

Aida Telegrafi, Bryn D Webb, Sarah M Robbins, Carlos E Speck-Martins, David FitzPatrick, Leah Fleming, Richard Redett, Andreas Dufke, Gunnar Houge, Jeske J T van Harssel and 10 more

2 registry-linked trialsOpen access · greenAbstract readCase Reports
In one paragraph

Article in American journal of medical genetics. Part A, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to 2 registered trials, which are not on this map. Cited by 18 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
18citing papers in PubMed, 1 pooled it
2.6field-weighted citation impact, top 10% 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.

NCT02055248 completednot on this map

Study on Moebius Syndrome and Other Congenital Facial Weakness Disorders

TypeobservationalSponsorNational Human Genome Research Institute (NHGRI)Ran2014 to 2020Enrolled207ConditionsBrain Disorders, Birth Defects, Craniofacial Differences
NCT03059420 recruitingnot on this map

Genetic Studies of Strabismus, Congenital Cranial Dysinnervation Disorders (CCDDs), and Their Associated Anomalies

TypeobservationalSponsorBoston Children's HospitalRan2004 to 2030Enrolled20,000ConditionsCongenital Fibrosis of Extraocular Muscles, Duane Retraction Syndrome, Duane Radial Ray Syndrome, Mobius Syndrome
3 · Its place in the literature

Who cites it

18 citing papers in PubMed, 1 synthesis or guideline pooled it, 37 citations in OpenAlex.

  1. Guideline
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  9. CaHandbook of experimental pharmacology · 2023
    Article
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  15. Skeletal muscle CaPflugers Archiv : European journal of physiology · 2020
    Review
  16. Duplex signaling by CaM and Stac3 enhances CaThe Journal of general physiology · 2018
    Article
  17. Article
  18. Article
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

20 authors at 11 institutions in 7 countries.

Aida TelegrafiGeneDx, Gaithersburg, Maryland.ORCID http://orcid.org/0000-0002-4211-5928
Bryn D WebbDepartment of Genetics and Genomic Sciences, Icahn School of Medicine at Mount Sinai, New York, New York.ORCID http://orcid.org/0000-0001-6174-4677
Sarah M RobbinsMcKusick-Nathans Institute of Genetic Medicine, Johns Hopkins University School of Medicine, Baltimore, Maryland.
Carlos E Speck-MartinsMedical Genetic Unit, SARAH Network of Rehabilitation Hospitals, Brasilia-DF, Brazil.
David FitzPatrickHuman Genetics Unit, Medical and Developmental Genetics, University of Edinburgh Western General Hospital, Edinburgh, United Kingdom.
Leah FlemingMcKusick-Nathans Institute of Genetic Medicine, Johns Hopkins University School of Medicine, Baltimore, Maryland.ORCID http://orcid.org/0000-0002-6742-7223
Richard RedettDepartment of Plastic & Reconstructive Surgery, Johns Hopkins Hospital University School of Medicine, Baltimore, Maryland.
Andreas DufkeInstitute of Medical Genetics and Applied Genomics, University of Tübingen, Tübingen, Germany.
Gunnar HougeCenter for Medical Genetics and Molecular Medicine, Haukeland University Hospital, Bergen, Norway.ORCID http://orcid.org/0000-0002-6102-1513
Jeske J T van HarsselDepartment of Clinical Genetics, University Medical Center, University of Utrecht, Utrecht, The Netherlands.
Alain VerloesDepartment of Genetics-Hospital Robert DEBRE, Paris, France.
Angela RoblesDr. Angela Robles Pediatrics Private Practice, San Sebastian, Puerto Rico.
Irini ManoliMedical Genomics and Metabolic Genetics Branch, National Human Genome Research Institute, National Institutes of Health, Bethesda, Maryland.
Elizabeth C EngleDepartment of Neurology, Boston Children's Hospital and Harvard Medical School, Boston, Massachusetts.
Moebius Syndrome Research Consortium
Ethylin W JabsDepartment of Genetics and Genomic Sciences, Icahn School of Medicine at Mount Sinai, New York, New York.
David ValleMcKusick-Nathans Institute of Genetic Medicine, Johns Hopkins University School of Medicine, Baltimore, Maryland.
John CareyDepartment of Pediatrics, University of Utah, Salt Lake City, Utah.
Julie E Hoover-FongMcKusick-Nathans Institute of Genetic Medicine, Johns Hopkins University School of Medicine, Baltimore, Maryland.ORCID http://orcid.org/0000-0002-1242-5626
Nara L M SobreiraMcKusick-Nathans Institute of Genetic Medicine, Johns Hopkins University School of Medicine, Baltimore, Maryland.
Johns Hopkins University · USIcahn School of Medicine at Mount Sinai · USBoston Children's Hospital · USClinical Research Puerto Rico · PRHaukeland University Hospital · NOHôpital Robert-Debré · FRNational Institutes of Health · USSarah Network of Rehabilitation Hospitals · BRUniversity of Tübingen · DEUniversity of Utah · USWestern General Hospital · GB

Funding

University of Utah Center for Clinical and Translational ScienceUL1TR001067 · NCATS · UNIVERSITY OF UTAH · PI DERE, WILLARD H., HESS, RACHEL · 2013 to 2017
$19.8M
Baylor-Johns Hopkins Center for Mendelian GeneticsU54HG006542 · NHGRI · JOHNS HOPKINS UNIVERSITY · PI VALLE, DAVID · 2012 to 2015
$15.6M
Baylor-Johns Hopkins Center for Mendelian GeneticsUM1HG006542 · NHGRI · JOHNS HOPKINS UNIVERSITY · PI VALLE, DAVID · 2016 to 2020
$14.5M
TRAINING GRANT IN GENETICST32GM007748 · NIGMS · HARVARD UNIVERSITY (MEDICAL SCHOOL) · PI Anne O'Donnell-Luria, Louise Wilkins-Haug · 1985 to 2026
$12.0M
Predoctoral Training Program in Human GeneticsT32GM007814 · NIGMS · JOHNS HOPKINS UNIVERSITY · PI VALLE, DAVID · 1985 to 2021
$10.6M
Mouse Neurodevelopmental Behavior CoreU54HD090255 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI KUNKEL, LOUIS M · 2016 to 2020
$5.2M
Genetics of Moebius syndrome and other congenital facial weakness disordersZIAHG200389 · NHGRI · NATIONAL HUMAN GENOME RESEARCH INSTITUTE · PI COLLINS, FRANCIS S. · 2013 to 2024
$4.9M
Birth Defects: Moebius syndrome and related facial weakness disordersU01HD079068 · NICHD · ICAHN SCHOOL OF MEDICINE AT MOUNT SINAI · PI BROOKS, BRIAN PATRICK, ENGLE, ELIZABETH C. · 2014 to 2016
$1.1M
NCATS NIH HHS UL1 TR001067NHGRI NIH HHS U54 HG006542NHGRI NIH HHS UM1 HG006542NICHD NIH HHS U01 HD079068NICHD NIH HHS U54 HD090255NIGMS NIH HHS T32 GM007748NIGMS NIH HHS T32 GM007814
6 · The paper itself

Abstract

Horstick et al. (2013) previously reported a homozygous p.Trp284Ser variant in STAC3 as the cause of Native American myopathy (NAM) in 5 Lumbee Native American families with congenital hypotonia and weakness, cleft palate, short stature, ptosis, kyphoscoliosis, talipes deformities, and susceptibility to malignant hyperthermia (MH). Here we present two non-Native American families, who were found to have STAC3 pathogenic variants. The first proband and her affected older sister are from a consanguineous Qatari family with a suspected clinical diagnosis of Carey-Fineman-Ziter syndrome (CFZS) based on features of hypotonia, myopathic facies with generalized weakness, ptosis, normal extraocular movements, cleft palate, growth delay, and kyphoscoliosis. We identified the homozygous c.851G>C;p.Trp284Ser variant in STAC3 in both sisters. The second proband and his affected sister are from a non-consanguineous, Puerto Rican family who was evaluated for a possible diagnosis of Moebius syndrome (MBS). His features included facial and generalized weakness, minimal limitation of horizontal gaze, cleft palate, and hypotonia, and he has a history of MH. The siblings were identified to be compound heterozygous for STAC3 variants c.851G>C;p.Trp284Ser and c.763_766delCTCT;p.Leu255IlefsX58. Given the phenotypic overlap of individuals with CFZS, MBS, and NAM, we screened STAC3 in 12 individuals diagnosed with CFZS and in 50 individuals diagnosed with MBS or a congenital facial weakness disorder. We did not identify any rare coding variants in STAC3. NAM should be considered in patients presenting with facial and generalized weakness, normal or mildly abnormal extraocular movement, hypotonia, cleft palate, and scoliosis, particularly if there is a history of MH.

Indexed as

MutationAdaptor Proteins, Signal TransducingAdolescentAdultChildFemaleHumansMaleMobius SyndromeMuscular DiseasesPedigreePierre Robin SyndromePrognosisYoung AdultAdaptor Proteins, Signal TransducingStac3 protein, humanCarey-Fineman-Ziter syndromecleft palateMoebius syndromeMyopathyNative American Myopathyp.Trp284Ser variantPuerto RicanQatar

Identifiers

PMID28777491
PMCPMC5843189
OpenAlexW2742789743

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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.