Evidence map›Paper›PMID 37840096›Full record

ArticleJournal of autism and developmental disorders2024

Sensory Symptoms and Signs of Hyperarousal in Individuals with Fragile X Syndrome: Findings from the FORWARD Registry and Database Multisite Study.

Ave M Lachiewicz, Tracy M Stackhouse, Kristin Burgess, Debra Burgess, Howard F Andrews, Tse-Hwei Choo, Walter E Kaufmann, Sharon A Kidd

Open access · hybridAbstract readMulticenter Study
In one paragraph

Article in Journal of autism and developmental disorders, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.

0numbers the graph read from it
0cells of the map it votes in
10citing papers in PubMed
3.4field-weighted citation impact, top 7% 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

10 citing papers in PubMed, 11 citations in OpenAlex.

  1. Profile of Behavioral Comorbidities in Children With Fragile X Syndrome.Journal of autism and developmental disorders · 2026
    Article
  2. Article
  3. Article
  4. Article
  5. Article
  6. Article
  7. Article
  8. Hyper-extralemniscal model of Fragile X syndrome.Cerebral cortex (New York, N.Y. : 1991) · 2025
    Article
  9. Article
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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

8 authors at 7 institutions in 1 country.

Ave M LachiewiczDepartment of Pediatrics, Duke University Health System, Durham, NC, USA. ave.lachiewicz@duke.edu.ORCID http://orcid.org/0000-0002-2934-3088
Tracy M StackhouseDevelopmental FX, Denver, CO, USA.ORCID http://orcid.org/0000-0001-6540-803X
Kristin BurgessNorth Carolina Fragile X Foundation, Durham, NC, USA.
Debra BurgessDepartment of Pediatrics, Duke University Health System, Durham, NC, USA.
Howard F AndrewsDepartments of Psychiatry and Biostatistics, Mailman School of Public Health, Columbia University, Irving Medical Center, New York, NY, USA.ORCID http://orcid.org/0000-0002-7726-0551
Tse-Hwei ChooDivision of Mental Health Data Science, New York State Psychiatric Institute, New York, NY, USA.ORCID http://orcid.org/0000-0001-5068-9799
Walter E KaufmannDepartment of Human Genetics, Emory University School of Medicine, Atlanta, GA, USA.ORCID http://orcid.org/0000-0002-8561-8453
Sharon A KiddDepartment of Pediatrics, University of California San Francisco, San Francisco, CA, USA.ORCID http://orcid.org/0000-0002-7562-8682
Duke University Health System · USColumbia University Irving Medical Center · USEmory University · USFX Palo Alto Laboratory · USNational Fragile X Foundation · USNew York State Office of Mental Health · USUniversity of California, San Francisco · US

Funding

Using Longitudinal Data to Characterize the Natural History of Fragile X Syndrome--Component AU01DD001189 · DD · INSTITUTE FOR BASIC RES IN DEV DISABIL · PI VELINOV, MILEN T · 2015 to 2019
$1.7M
CDC HHS U01DD000231CDC HHS U01DD000753CDC HHS U01DD001189NCBDD CDC HHS U01 DD001189
6 · The paper itself

Abstract

This study was designed to increase our understanding about characteristics and the impact of sensory symptoms (SS) and signs of hyperarousal (HA) in individuals with fragile X syndrome (FXS) from childhood through early adulthood and by gender. Data derived from the Fragile X Online Registry With Accessible Research Database (FORWARD), a natural history study of FXS, were analyzed using descriptive statistics and multivariate linear and logistic regression models to examine SS and signs of HA, their impact on behavioral regulation and limitations on the subject/family. The sample (N = 933) consisted of 720 males and 213 females. More males were affected with SS (87% vs. 68%) and signs of HA (92% vs. 79%). Subjects who were endorsed as having a strong sensory response had more comorbidities, including behavioral problems. The predominant SS was difficulty with eye gaze that increased with age in both genders. As individuals age, there was less use of non-medication therapies, such as occupational therapy (OT)/physical therapy (PT), but there was more use of psychopharmacological medications and investigational drugs for behaviors. Multiple regression models suggested that endorsing SS and signs of HA was associated with statistically significantly increased ABC-C-I subscale scores and limited participation in everyday activities. This study improves our understanding of SS and signs of HA as well as their impact in FXS. It supports the need for more research regarding these clinical symptoms, especially to understand how they contribute to well-known behavioral concerns.

Indexed as

Fragile X SyndromeRegistriesAdolescentAdultArousalChildChild, PreschoolComorbidityDatabases, FactualFemaleHumansMaleYoung AdultFragile X syndromeHyperarousalSensory symptoms

Identifiers

PMID37840096
PMCPMC11461590
OpenAlexW4387651515

What Socratic 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 Socratic graph.