Evidence map›Paper›PMID 31104364›Full record

ArticleMolecular genetics & genomic medicine2019

Cognitive and behavioral improvement in adults with fragile X syndrome treated with metformin-two cases.

Dragana Protic, Elber Y Aydin, Flora Tassone, Maria M Tan, Randi J Hagerman, Andrea Schneider

Registry-linked trialAbstract readCase Reports
In one paragraph

Article in Molecular genetics & genomic medicine, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT04930471 (A Multi-center, Prospective, Cohort Study to Elucidate the Effects of Metformin Treatment on Steroid Hormones and Social Behavior. Linking Autistic Behaviorial Symptoms to Changes in Steroid Hormone Availability), which is not on this map. Cited by 22 papers.

0numbers the graph read from it
0cells of the map it votes in
22citing papers in PubMed
–field-weighted citation impact
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.

NCT04930471 unknown statusnot on this mapstarted 2021, after this paper: background citation

A Multi-center, Prospective, Cohort Study to Elucidate the Effects of Metformin Treatment on Steroid Hormones and Social Behavior. Linking Autistic Behaviorial Symptoms to Changes in Steroid Hormone Availability

TypeobservationalSponsorInsel Gruppe AG, University Hospital BernRan2021 to 2023Enrolled45ConditionsAutistic DisorderArmsMetformin
3 · Its place in the literature

Who cites it

22 citing papers in PubMed.

  1. Trial
  2. Review
  3. State-of-the-art therapies for fragile X syndrome.Developmental medicine and child neurology · 2024
    Review
  4. Review
  5. Article
  6. Article
  7. Genes · 2022
    Review
  8. Article
  9. Article
  10. Article
  11. Fragile X Syndrome: From Molecular Aspect to Clinical Treatment.International journal of molecular sciences · 2022
    Review
  12. Article
  13. Dysregulation of BMP, Wnt, and Insulin Signaling in Fragile X Syndrome.Frontiers in cell and developmental biology · 2022
    Review
  14. An Update on Psychopharmacological Treatment of Autism Spectrum Disorder.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2022
    Review
  15. Transcriptional Regulation ofBiomolecules · 2021
    Article
  16. Article
  17. Article
  18. Article
  19. Article
  20. 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

6 authors.

Dragana ProticMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California Davis, Sacramento, California.ORCID 0000-0002-2137-5405
Elber Y AydinMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California Davis, Sacramento, California.
Flora TassoneMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California Davis, Sacramento, California.
Maria M TanMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California Davis, Sacramento, California.
Randi J HagermanMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California Davis, Sacramento, California.
Andrea SchneiderMedical Investigation of Neurodevelopmental Disorders (MIND) Institute, University of California Davis, Sacramento, California.ORCID 0000-0002-4674-7244

Funding

UC Davis Clinical and Translational Science CenterUL1TR001860 · NCATS · UNIVERSITY OF CALIFORNIA AT DAVIS · PI KENYON, NICHOLAS J., LYLES, COURTNEY REES · 2016 to 2025
$47.3M
Research Project: Pathologic Significance of Maternal AutoantibodiesP50HD103526 · NICHD · UNIVERSITY OF CALIFORNIA AT DAVIS · PI JUDY A. VAN DE WATER · 2020 to 2026
$9.7M
Rodent Behavior CoreU54HD079125 · NICHD · UNIVERSITY OF CALIFORNIA AT DAVIS · PI ABBEDUTO, LEONARD J. · 2013 to 2019
$8.5M
NCATS NIH HHS UL1 TR001860NICHD NIH HHS P50 HD103526NICHD NIH HHS U54 HD079125NIH HHS UL1 TR001860
6 · The paper itself

Abstract

backgroundThe majority of individuals with fragile X syndrome (FXS) have intellectual disability, behavioral problems, autism, and language deficits. IQ typically declines with age in boys with the full mutation. The results of preclinical studies demonstrated that metformin, a biguanide used to treat type 2 diabetes, rescues multiple phenotypes of FXS in both Drosophila and mouse models. Preliminary studies of patients with FXS demonstrated improvements in behavior.

methodsHere, we present two cases of individuals who have been treated with metformin clinically for one year.

resultsBoth patients demonstrated significant cognitive and behavioral improvements. They also improved eating habits and normalization of their weight percentiles.

conclusionMetformin may be a candidate drug for treatment of several types of symptoms in individuals with FXS.

Indexed as

AdultCognitionFragile X SyndromeHumansIntelligence TestsMaleMetforminMetformincognition improvementfragile X syndromemetformintargeted treatment

Identifiers

PMID31104364
PMCPMC6625129

What OpenQuestion holds

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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.