Frontiers in Psychology (Jun 2024)
Longitudinal follow-up of metformin treatment in Fragile X Syndrome
- Panhaneath Seng,
- Federica Alice Maria Montanaro,
- Federica Alice Maria Montanaro,
- Hazel Maridith Barlahan Biag,
- Hazel Maridith Barlahan Biag,
- Maria Jimena Salcedo-Arellano,
- Maria Jimena Salcedo-Arellano,
- Kyoungmi Kim,
- Kyoungmi Kim,
- Kyoungmi Kim,
- Matthew Dominic Ponzini,
- Matthew Dominic Ponzini,
- Flora Tassone,
- Flora Tassone,
- Andrea Schneider,
- Andrea Schneider,
- Leonard Abbeduto,
- Leonard Abbeduto,
- Angela John Thurman,
- Angela John Thurman,
- David Hessl,
- David Hessl,
- Francois V. Bolduc,
- Sebastien Jacquemont,
- Sebastien Jacquemont,
- Sarah Lippé,
- Sarah Lippé,
- Randi J. Hagerman,
- Randi J. Hagerman
Affiliations
- Panhaneath Seng
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Federica Alice Maria Montanaro
- Child and Adolescent Neuropsychiatry Unit, Bambino Gesù Children’s Hospital, IRCCS, Rome, Italy
- Federica Alice Maria Montanaro
- Department of Education, Psychology, Communication, University of Bari Aldo Moro, Bari, Italy
- Hazel Maridith Barlahan Biag
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Hazel Maridith Barlahan Biag
- Department of Pediatrics, University of California Davis School of Medicine, Sacramento, CA, United States
- Maria Jimena Salcedo-Arellano
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Maria Jimena Salcedo-Arellano
- Department of Pediatrics, University of California Davis School of Medicine, Sacramento, CA, United States
- Kyoungmi Kim
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Kyoungmi Kim
- Department of Public Health Sciences, University of California Davis School of Medicine, Sacramento, CA, United States
- Kyoungmi Kim
- Integrative Genetics and Genomics Graduate Group, University of California Davis, Davis, CA, United States
- Matthew Dominic Ponzini
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Matthew Dominic Ponzini
- Department of Public Health Sciences, University of California Davis School of Medicine, Sacramento, CA, United States
- Flora Tassone
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Flora Tassone
- Department of Biochemistry and Molecular Medicine, University of California Davis School of Medicine, Sacramento, CA, United States
- Andrea Schneider
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Andrea Schneider
- Department of Pediatrics, University of California Davis School of Medicine, Sacramento, CA, United States
- Leonard Abbeduto
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Leonard Abbeduto
- Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, United States
- Angela John Thurman
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Angela John Thurman
- Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, United States
- David Hessl
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- David Hessl
- Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA, United States
- Francois V. Bolduc
- Department of Pediatrics, Department of Medical Genetics, Women and Children Health Research Institute, University of Alberta, Edmonton, AB, Canada
- Sebastien Jacquemont
- 0CHU Sainte-Justine Research Center, Université de Montréal, Montreal, QC, Canada
- Sebastien Jacquemont
- 1Department of Pediatrics, University of Montreal, Montreal, QC, Canada
- Sarah Lippé
- 0CHU Sainte-Justine Research Center, Université de Montréal, Montreal, QC, Canada
- Sarah Lippé
- 2Department of Psychology, Université de Montréal, Montreal, QC, Canada
- Randi J. Hagerman
- MIND Institute, University of California Davis Health System, Sacramento, CA, United States
- Randi J. Hagerman
- Department of Public Health Sciences, University of California Davis School of Medicine, Sacramento, CA, United States
- DOI
- https://doi.org/10.3389/fpsyg.2024.1305597
- Journal volume & issue
-
Vol. 15
Abstract
IntroductionMetformin has been used as a targeted treatment to potentially improve cognition and slow the typical IQ decline that occurs during development among individuals with fragile X syndrome (FXS). In this follow-up study, we are following the trajectory of IQ and adaptive behavior changes over 1 to 3 years in individuals with FXS who are clinically treated with metformin in an open label trial.MethodIndividuals with FXS ages 6 to 25 years (mean 13.15 ± 5.50) and nonverbal IQ mean 57.69 (±15.46) were treated for 1–3 years (1.88 ± 0.63). They all had a baseline IQ test using the Leiter-III non-verbal cognitive assessment and the Vineland-III adaptive behavior assessment before the start of metformin. Repeat Leiter-III and Vineland-III were completed after at least 1 year of metformin (500–1,000 mg/dose given twice a day).ResultThere were no significant changes in non-verbal IQ or in the adaptive behavior measurements at FDR < 0.05. The findings thus far indicate that both IQ and adaptive behavior are stable over time, and we did not see a significant decline in either measure.ConclusionOverall, the small sample size and short follow-up duration limit the interpretation of the effects of metformin on cognitive development and adaptive functioning. There is individual variability but overall for the group there was no significant decline in IQ or adaptive behavior.
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