Evidence map›Paper›PMID 36140827›Full record

ArticleGenes2022

Observable Symptoms of Anxiety in Individuals with Fragile X Syndrome: Parent and Caregiver Perspectives.

Reymundo Lozano, Talia Thompson, Jayne Dixon-Weber, Craig A Erickson, Elizabeth Berry-Kravis, Sara Williams, Elizabeth Smith, Jean A Frazier, Hilary Rosselot, Cristan Farmer and 1 more

Open access · goldAbstract read
In one paragraph

Article in Genes, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
11citing papers in PubMed, 1 pooled it
3.9field-weighted citation impact, top 6% 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.

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

11 citing papers in PubMed, 1 synthesis or guideline pooled it, 23 citations in OpenAlex.

  1. Pooled it
  2. Article
  3. Article
  4. Article
  5. Article
  6. Article
  7. Article
  8. Article
  9. Article
  10. Article
  11. 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

11 authors at 8 institutions in 1 country.

Reymundo LozanoDepartment of Genetics and Genomic Sciences, Icahn School of Medicine at Mount Sinai, New York, NY 10029, USA.ORCID 0000-0002-5616-5854
Talia ThompsonDepartment of Pediatrics, University of Colorado School of Medicine, Aurora, CO 80045, USA.
Jayne Dixon-WeberNational Fragile X Foundation, Washington, DC 20005, USA.
Craig A EricksonDepartment of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.
Elizabeth Berry-KravisDepartment of Pediatrics, Rush University Medical Center, Chicago, IL 60612, USA.
Sara WilliamsDepartment of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.
Elizabeth SmithDepartment of Pediatrics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.ORCID 0000-0003-4661-0050
Jean A FrazierDepartment of Psychiatry, Chan Medical School, University of Massachusetts, Worcester, MA 01655, USA.
Hilary RosselotNational Fragile X Foundation, Washington, DC 20005, USA.
Cristan FarmerNeurodevelopmental and Behavioral Phenotyping Service, National Institute of Mental Health, Bethesda, MD 20892, USA.
David HesslMIND Institute, University of California Davis Medical Center, Sacramento, CA 95817, USA.
Cincinnati Children's Hospital Medical Center · USNational Fragile X Foundation · USIcahn School of Medicine at Mount Sinai · USNational Institute of Mental Health · USRush University Medical Center · USUniversity of California Davis Medical Center · USUniversity of Colorado Denver · USUniversity of Massachusetts Chan Medical School · US

Funding

FXTAS Key Molecular Pathways Converge with Other Neurodegenerative DisordersK01NS126736 · NINDS · ICAHN SCHOOL OF MEDICINE AT MOUNT SINAI · PI Reymundo Lozano · 2022 to 2026
$1.4M
NINDS NIH HHS 1K01NS126736-01NINDS NIH HHS K01 NS126736
6 · The paper itself

Abstract

Caregiver reports, clinical observations, and diagnostic assessments indicate that most individuals with fragile X syndrome experience high levels of chronic anxiety. However, anxiety is a challenging endpoint for outcome measurement in FXS because most individuals cannot reliably report internal emotional or body states. A comprehensive survey of the presence, frequency, and duration of anxiety-related symptoms and questions to elicit open-ended responses was completed by caregivers of 456 individuals with FXS, ages 2-81 years (87 female, 369 male) and 24 female and 2 male FXS self-advocates ages 15-66 years. Caregivers reported classic behavioral indicators of anxiety, such as avoidance, irritability, motor agitation, and physiological symptoms, as well as behavioral features in FXS such as repetitive behavior, aggression, and self-injury. Self-advocate accounts largely paralleled caregiver data. Factor analyses yielded four factors: (1) increased irritability, aggression, and self-injury; (2) increased physical movement, nervous activity, and restlessness; (3) physical and physiological features of anxiety; and (4) internalizing and gastrointestinal symptoms. Caregivers are capable of observing and reporting behaviors that are valid indicators of anxious states that are usually reported in self-report standardized assessments. These results support the development of an anxiety measure for FXS that minimizes problems with rater inference.

Indexed as

Fragile X SyndromeAdolescentAdultAgedAged, 80 and overAnxietyAnxiety DisordersCaregiversChildChild, PreschoolFemaleHumansMaleMiddle AgedParentsYoung AdultassessmentautismFMR1 geneintellectual disability

Identifiers

PMID36140827
PMCPMC9498703
OpenAlexW4296354900

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

Registered trials

None linked

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.