Metformin treatment in young children with fragile X syndrome
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Background: Metformin is a drug commonly used in individuals with type 2 diabetes, obesity, and impaired glucose tolerance. It has a strong safety profile in both children and adults. Studies utilizing the Drosophila model and knock out mouse model of fragile X syndrome (FXS) have found metformin to rescue memory, social novelty deficits, and neuroanatomical abnormalities. These studies provided preliminary evidence that metformin could be used as a targeted treatment for the cognitive and behavioral problems associated with FXS. Previously, a case series of children and adults with FXS treated with metformin demonstrated improvements in irritability, social responsiveness, language, and hyperactivity. Methods: Here, we present nine children with FXS between 2 and 7 years of age who were treated clinically with metformin and monitored for behavioral and metabolic changes. Results: Parent reports and developmental testing before and after metformin are presented. There were improvements in language development and behavior (such as lethargy and stereotypy) in most of the patients. Conclusion: These results support the need for a controlled trial of metformin in children with FXS under 7 years old whose brains are in a critical developmental window and thus may experience a greater degree of clinical benefit from metformin.
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Additional open label studies with metformin in children ages 2 to 7 years old with FXS showed improvements in behavior and development on the MSEL.
“Additional open label studies have been carried out with metformin including one in children ages 2 to 7 years old with FXS, and improvements were seen in behavior and development on the MSEL [110].”
From An Update on Psychopharmacological Treatment of Autism Spectrum Disorder (2022) · cites this paper as [110]
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Cites
Individual Comparisons by Ranking Methods
Wilcoxon, F. (1945) · Biometrics Bulletin · doi:10.2307/3001968 · Cited by 15,354 · see citing works
Bayley Scales of Infant and Toddler Development, Third Edition
Bayley, N. (2005) · PsycTESTS Dataset · doi:10.1037/t14978-000 · Cited by 1,200 · see citing works
Bolen, S. D., Feldman, L. S., Vassy, J. L., Wilson, L. M., Yeh, H., Marinopoulos, S. S., Wiley, C., Selvin, E., Wilson, R. F., Bass, E. B., et al. (2007) · Annals of Internal Medicine · doi:10.7326/0003-4819-147-6-200709180-00178 · Cited by 816 · see citing works
Mullen Scales of Early Learning: Ags Edition
Dumont, R., & Willis, J. O. (2008) · Encyclopedia of Special Education · doi:10.1002/9780470373699.speced1402 · Cited by 667 · see citing works
Bilousova, T. V., Dansie, L., Ngo, M., Aye, J., Charles, J. R., Ethell, D. W., & Ethell, I. M. (2009) · Journal of Medical Genetics · doi:10.1136/jmg.2008.061796 · Cited by 465 · see citing works
Bodmer, M., Meier, C., Krähenbühl, S., Jick, S. S., & Meier, C. R. (2008) · Diabetes Care · doi:10.2337/dc08-1171 · Cited by 422 · see citing works
Effect of Metformin in Pediatric Patients With Type 2 Diabetes
Jones, K. L., Arslanian, S., Peterokova, V. A., Park, J.-S., & Tomlinson, M. J. (2002) · Diabetes Care · doi:10.2337/diacare.25.1.89 · Cited by 392 · see citing works
Vitamin B12 Deficiency: Recognition and Management
Cyran, E. M. (2002) · Primary Care Case Reviews · doi:10.1097/00129300-200206000-00001 · Cited by 309 · see citing works
Leigh, M. J. S., Nguyen, D. V., Mu, Y., Winarni, T. I., Schneider, A., Chechi, T., Polussa, J., Doucet, P., Tassone, F., Rivera, S. M., et al. (2013) · Journal of Developmental & Behavioral Pediatrics · doi:10.1097/dbp.0b013e318287cd17 · Cited by 298 · see citing works
Self-citationKlein, D. J., Cottingham, E. M., Sorter, M. T., BARTON, B. A., & Morrison, J. A. (2006) · American Journal of Psychiatry · doi:10.1176/ajp.2006.163.12.2072 · Cited by 271 · see citing works
Vitamin B12 Deficiency: Recognition and Management.
Langan, R. C., & Goodbred, A. J. (2017) · PubMed · Cited by 243 · see citing works
Metformin ameliorates core deficits in a mouse model of fragile X syndrome
Gantois, I., Khoutorsky, A., Popić, J., Aguilar‐Valles, A., Freemantle, E., Cao, R., Sharma, V. K., Pooters, T., Nagpal, A., Skalecka, A., et al. (2017) · Nature Medicine · doi:10.1038/nm.4335 · Cited by 228 · see citing works
Altered mTOR signaling and enhanced CYFIP2 expression levels in subjects with fragile X syndrome
Hoeffer, C. A., Sanchez, E., Hagerman, R. J., Mu, Y., Nguyen, D. V., Wong, H., Whelan, A. M., Zukin, R. S., Klann, E., & Tassone, F. (2012) · Genes, Brain and Behavior · doi:10.1111/j.1601-183x.2012.00768.x · Cited by 198 · see citing works
Self-citationArbaclofen in fragile X syndrome: results of phase 3 trials
Berry-Kravis, E., Hagerman, R., Visootsak, J., Budimirovic, D., Kaufmann, W. E., Cherubini, M., Zarevics, P., Walton-Bowen, K., Wang, P., Bear, M. F., et al. (2017) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-016-9181-6 · Cited by 183 · see citing works
Self-citationSansone, S. M., Widaman, K. F., Hall, S. S., Reiss, A. L., Lightbody, A. A., Kaufmann, W. E., Berry‐Kravis, E., Lachiewicz, A. M., Brown, E. C., & Hessl, D. R. (2011) · Journal of Autism and Developmental Disorders · doi:10.1007/s10803-011-1370-2 · Cited by 177 · see citing works
The Prader-Willi phenotype of fragile X syndrome
Nowicki, S. T., Tassone, F., Ono, M. Y., Ferranti, J., Croquette, M. F., Goodlin-Jones, B., & Hagerman, R. J. (2007) · Journal of Developmental & Behavioral Pediatrics · doi:10.1097/01.dbp.0000267563.18952.c9 · Cited by 174 · see citing works
Self-citationAttention, Development, and Psychopathology
Merrill, E. C., & Atwell, J. A. (1998) · American Journal on Mental Retardation · doi:10.1352/0895-8017(1998)103 · Cited by 170 · see citing works
Metformin for Obesity in Children and Adolescents: A Systematic Review
Park, M. H., Kinra, S., Ward, K. J., White, B., & Viner, R. (2009) · Diabetes Care · doi:10.2337/dc09-0258 · Cited by 165 · see citing works
Seizures in Fragile X Syndrome: Characteristics and Comorbid Diagnoses
Berry-Kravis, E., Raspa, M., Loggin-Hester, L., Bishop, E., Holiday, D., & Bailey, D. B. (2010) · American Journal on Intellectual and Developmental Disabilities · doi:10.1352/1944-7558-115.6.461 · Cited by 155 · see citing works
He, C. X., Cantu, D. A., Mantri, S. S., Zeiger, W., Goel, A., & Portera‐Cailliau, C. (2017) · Journal of Neuroscience · doi:10.1523/jneurosci.0651-17.2017 · Cited by 140 · see citing works
Rotschafer, S. E., Trujillo, M. S., Dansie, L. E., Ethell, I. M., & Razak, K. A. (2012) · Brain Research · doi:10.1016/j.brainres.2011.12.041 · Cited by 130 · see citing works
MMP9: A novel function in synaptic plasticity
Dziembowska, M., & Włodarczyk, J. (2012) · The International Journal of Biochemistry & Cell Biology · doi:10.1016/j.biocel.2012.01.023 · Cited by 125 · see citing works
Metformin as targeted treatment in fragile X syndrome
Dy, A. B. C., Tassone, F., Eldeeb, M., Salcedo‐Arellano, M. J., Tartaglia, N., & Hagerman, R. (2018) · Clinical Genetics · doi:10.1111/cge.13039 · Cited by 124 · see citing works
Self-citationZhao, R., Xu, X., Xu, F., Zhang, W.-L., Zhang, W.-L., Liu, L.-M., & Wang, W. (2014) · Biochemical and Biophysical Research Communications · doi:10.1016/j.bbrc.2014.04.130 · Cited by 123 · see citing works
Anagnostou, E., Aman, M. G., Handen, B. L., Sanders, K. B., Shui, A., Hollway, J. A., Brian, J., Arnold, L. E., Capano, L., Hellings, J. A., et al. (2016) · JAMA Psychiatry · doi:10.1001/jamapsychiatry.2016.1232 · Cited by 123 · see citing works
Greiss Hess, L., Fitzpatrick, S. E., Nguyen, D. V., Chen, Y., Gaul, K. N., Schneider, A., Lemons Chitwood, K., Eldeeb, M. A. A. A., Polussa, J., Hessl, D., et al. (2016) · Journal of Developmental & Behavioral Pediatrics · doi:10.1097/dbp.0000000000000334 · Cited by 122 · see citing works
Self-citationESFAHANIAN, N., SHAKIBA, Y., NIKBIN, B., SORAYA, H., MALEKI-DIZAJI, N., GHAZI-KHANSARI, M., & GARJANI, A. (2012) · Molecular Medicine Reports · doi:10.3892/mmr.2012.753 · Cited by 120 · see citing works
Monyak, R. E., Emerson, D., Schoenfeld, B. P., Zheng, X., Chambers, D. B., Rosenfelt, C., Langer, S., Hinchey, P., Choi, C. H., McDonald, T. V., et al. (2017) · Molecular Psychiatry · doi:10.1038/mp.2016.51 · Cited by 100 · see citing works
Longitudinal IQ changes in fragile X males
Hagerman, R. J., Schreiner, R. A., Kemper, M. B., Wittenberger, M. D., Zahn, B., & Habicht, K. (1989) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320330422 · Cited by 89 · see citing works
Self-citationMetformin for treatment of fragile X syndrome and other neurological disorders
Gantois, I., Popic, J., Khoutorsky, A., & Sonenberg, N. (2019) · Annual Review of Medicine · doi:10.1146/annurev-med-081117-041238 · Cited by 87 · see citing works
Declining IQs of young males with the fragile X syndrome.
Lachiewicz, A. M., Gullion, C. M., Spiridigliozzi, G. A., & Aylsworth, A. S. (1987) · PubMed · Cited by 86 · see citing works
Developmental Implications of Changing Trajectories of IQ in Males with Fragile X Syndrome
Hodapp, R. M., Dykens, E. M., Hagerman, R. J., Schreiner, R. A., Lachiewicz, A. M., & Leckman, J. F. (1990) · Journal of the American Academy of Child & Adolescent Psychiatry · doi:10.1097/00004583-199003000-00009 · Cited by 84 · see citing works
Self-citationFarmer, C., Golden, C., & Thurm, A. E. (2015) · Child Neuropsychology · doi:10.1080/09297049.2015.1020775 · Cited by 77 · see citing works
Long-Term Efficacy of Metformin Therapy in Nonobese Individuals With Type 2 Diabetes
Ong, C. R., Molyneaux, L. M., Constantino, M., Twigg, S. M., & Yue, D. K. (2006) · Diabetes Care · doi:10.2337/dc06-0827 · Cited by 76 · see citing works
Li, W., Li, N.-P., Song, D., Rong, J., Qian, A., & Li, X. (2017) · International Journal of Molecular Medicine · doi:10.3892/ijmm.2017.2929 · Cited by 76 · see citing works
Trajectories and Predictors of the Development of Very Young Boys with Fragile X Syndrome
Roberts, J. E., Mankowski, J., Sideris, J. H., Goldman, B. D., Hatton, D. D., Mirrett, P. L., Baranek, G. T., Reznick, J. S., Long, A. C. J., & Bailey, D. B. (2008) · Journal of Pediatric Psychology · doi:10.1093/jpepsy/jsn129 · Cited by 75 · see citing works
Chronic metformin treatment facilitates seizure termination
Yang, Y., Zhu, B., Zheng, F., Li, Y., Zhang, Y., Hu, Y., & Wang, X. (2017) · Biochemical and Biophysical Research Communications · doi:10.1016/j.bbrc.2017.01.157 · Cited by 66 · see citing works
Mehrabi, S., Sanadgol, N., Barati, M., Shahbazi, A., Vahabzadeh, G., Barzroudi, M., Seifi, M., Gholipourmalekabadi, M., & Golab, F. (2017) · Metabolic Brain Disease · doi:10.1007/s11011-017-0132-z · Cited by 62 · see citing works
Infant Development in Fragile X Syndrome: Cross-Syndrome Comparisons
Roberts, J. E., McCary, L. M., Shinkareva, S. V., & Bailey, D. B. (2016) · Journal of Autism and Developmental Disorders · doi:10.1007/s10803-016-2737-1 · Cited by 44 · see citing works
Growth of Expressive Syntax in Children With Fragile X Syndrome
Komesidou, R., Brady, N. C., Fleming, K., Esplund, A., & Warren, S. F. (2017) · Journal of Speech Language and Hearing Research · doi:10.1044/2016_jslhr-l-15-0360 · Cited by 43 · see citing works
AlOlaby, R. R., Sweha, S. R., Silva, M., Durbin-Johnson, B. P., Yrigollen, C. M., Pretto, D. I., Hagerman, R. J., & Tassone, F. (2017) · Brain and Development · doi:10.1016/j.braindev.2017.01.012 · Cited by 41 · see citing works
Self-citationPeriventricular heterotopia in fragile X syndrome
Moro, F., Pisano, T., Bernardina, B. D., Polli, R., Murgia, A., Zoccante, L., Darra, F., Battaglia, A., Pramparo, T., Zuffardi, O., et al. (2006) · Neurology · doi:10.1212/01.wnl.0000230223.51595.99 · Cited by 39 · see citing works
Grefer, M. L., Flory, K., Cornish, K., Hatton, D., & Roberts, J. E. (2015) · Journal of Intellectual Disability Research · doi:10.1111/jir.12226 · Cited by 31 · see citing works
Longitudinal trajectories of aberrant behavior in fragile X syndrome
Hustyi, K. M., Hall, S. S., Jo, B., Lightbody, A. A., & Reiss, A. L. (2014) · Research in Developmental Disabilities · doi:10.1016/j.ridd.2014.07.003 · Cited by 28 · see citing works
The neurobiology of the Prader-Willi phenotype of fragile X syndrome
Muzar, Z., Lozano, R., Kolevzon, A. L., & Hagerman, R. J. (2016) · Intractable & Rare Diseases Research · doi:10.5582/irdr.2016.01082 · Cited by 22 · see citing works
Self-citationLee, M., Chang, H., Wu, H.-H., Weng, H., & Chen, C. (2005) · Epilepsia · doi:10.1111/j.1528-1167.2005.66204.x · Cited by 19 · see citing works
Long‐term potentiation in the hippocampus of fragile X knockout mice
Godfraind, J., Reyniers, E., Boulle, K. D., D’Hooge, R., Deyn, P. P. D., Bakker, C. E., Oostra, B. A., Kooy, R. F., & Willems, P. J. (1996) · American Journal of Medical Genetics · doi:10.1002/(sici)1096-8628(19960809)64:2 · Cited by 16 · see citing works
Self-citationVitamin B12 deficiency: an unusual cause for recurrent generalised seizures with pancytopaenia
Naha, K., Dasari, S., Vivek, G., & Prabhu, M. M. (2012) · BMJ Case Reports · doi:10.1136/bcr-2012-006632 · Cited by 14 · see citing works
Early Development in Fragile X Syndrome: Implications for Developmental Screening
Bailey, D., Hazlett, H., Roberts, J. E., & Wheeler, A. C. (2011) · International review of research in developmental disabilities · doi:10.1016/b978-0-12-374478-4.00004-6 · Cited by 5 · see citing works
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Aishworiya, R., Valica, T., Hagerman, R., & Restrepo, B. (2022) · Neurotherapeutics · doi:10.1007/s13311-022-01183-1 · Cited by 233 · see citing works
Self-citationFragile X Syndrome: From Molecular Aspect to Clinical Treatment
Protic, D. D., Aishworiya, R., Salcedo‐Arellano, M. J., Tang, S. J., Milisavljevic, J., Mitrovic, F., Hagerman, R. J., & Budimirovic, D. B. (2022) · International Journal of Molecular Sciences · doi:10.3390/ijms23041935 · Cited by 89 · see citing works
Self-citationEnergy metabolism in childhood neurodevelopmental disorders
Oyarzábal, A., Musokhranova, U., Barros, L. F., & García‐Cazorla, À. (2021) · EBioMedicine · doi:10.1016/j.ebiom.2021.103474 · Cited by 76 · see citing works
Liu, D., Nanclares, C., Simbriger, K., Fang, K., Lorsung, E., Le, N., Amorim, I. S., Chalkiadaki, K., Pathak, S. S., Li, J., et al. (2023) · Molecular Psychiatry · doi:10.1038/s41380-022-01499-6 · Cited by 56 · see citing works
Abbeduto, L., Berry‐Kravis, E., Sterling, A. M., Sherman, S. L., Edgin, J. O., McDuffie, A. S., Hoffmann, A., Hamilton, D., Nelson, M. N., Aschkenasy, J. R., et al. (2020) · Journal of Neurodevelopmental Disorders · doi:10.1186/s11689-020-09313-6 · Cited by 51 · see citing works
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Dong, J., Yin, J., Li, D., Zhu, C., Ye, J., & Pan, Y.-Q. (2020) · Scientific Reports · doi:10.1038/s41598-020-77370-x · Cited by 38 · see citing works
The safety and efficacy of metformin in fragile X syndrome: An open-label study
Proteau-Lemieux, M., Lacroix, A., Galarneau, L., Corbin, F., Lepage, J., & Çaku, A. (2021) · Progress in Neuro-Psychopharmacology and Biological Psychiatry · doi:10.1016/j.pnpbp.2021.110307 · Cited by 35 · see citing works
Chen, J., Zhou, T., Guo, A.-M., Chen, W., Lin, D., Liu, Z.-Y., & Fei, E. (2020) · Biology · doi:10.3390/biology9110359 · Cited by 27 · see citing works
Salcedo‐Arellano, M. J., Cabal-Herrera, A. M., Punatar, R., Clark, C. J., Romney, C. A., & Hagerman, R. J. (2020) · Neurotherapeutics · doi:10.1007/s13311-020-00968-6 · Cited by 30 · see citing works
Self-citationWong, H., Hooper, A. W. M., Niibori, Y., Lee, S. J., Hategan, L. A., Zhang, L., Karumuthil‐Melethil, S., Till, S. M., Kind, P. C., Danos, O., et al. (2020) · Neurobiology of Disease · doi:10.1016/j.nbd.2020.105118 · Cited by 27 · see citing works
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Protic, D. D., & Hagerman, R. J. (2024) · Developmental Medicine & Child Neurology · doi:10.1111/dmcn.15885 · Cited by 24 · see citing works
Self-citationMetformin Enhances Excitatory Synaptic Transmission onto Hippocampal CA1 Pyramidal Neurons
Chen, W., Chen, J., Liu, Z., Luo, B., Zhou, T. G., & Fei, E. (2020) · Brain Sciences · doi:10.3390/brainsci10100706 · Cited by 22 · see citing works
Sudhahar, S., Özer, B., Chang, J., Chadwick, W., O’Donovan, D., Campbell, A., Tulip, E., Thompson, N. T., & Roberts, I. (2024) · Nature Communications · doi:10.1038/s41467-024-50024-6 · Cited by 20 · see citing works
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Acero-Garcés, D. O., Saldarriaga, W., Cabal-Herrera, A. M., Cerón, C. A. R., & Hagerman, R. J. (2023) · Colombia medica · doi:10.25100/cm.v54i2.5089 · Cited by 14 · see citing works
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Johnson, D., Clark, C. J., & Hagerman, R. J. (2023) · Advances in neurobiology · doi:10.1007/978-3-031-21054-9_10 · Cited by 10 · see citing works
Self-citationMbachu, C. N. P., Mbachu, I. I., & Hagerman, R. J. (2024) · Genes · doi:10.3390/genes15060683 · Cited by 10 · see citing works
Self-citationFragile X Syndrome and Targeted Treatments
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Self-citationGasser, B. A., Kurz, J., Buerki, S., & Mohaupt, M. G. (2022) · Life · doi:10.3390/life12070998 · Cited by 7 · see citing works
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van der Lei, M. B., & Kooy, R. F. (2025) · Biomedicines · doi:10.3390/biomedicines13040805 · Cited by 6 · see citing works
Raspa, M., Wheeler, A., Okoniewski, K. C., Edwards, A., & Scott, S. (2022) · Journal of Developmental & Behavioral Pediatrics · doi:10.1097/dbp.0000000000001134 · Cited by 6 · see citing works
Arezoumand, K. S., Roberts, C.-T., & Rastegar, M. (2024) · Biomolecules · doi:10.3390/biom14040505 · Cited by 6 · see citing works
The Use of Peptides in the Treatment of Fragile X Syndrome: Challenges and Opportunities
Romagnoli, A., & Marino, D. D. (2021) · Frontiers in Psychiatry · doi:10.3389/fpsyt.2021.754485 · Cited by 7 · see citing works
Life, B. E., Bettio, L. E. B., Gantois, I., Christie, B. R., & Leavitt, B. R. (2023) · Current Research in Neurobiology · doi:10.1016/j.crneur.2023.100094 · Cited by 5 · see citing works
Gasser, B. A., Calin, A.-E., Escher, G., Kurz, J., Emmenegger, A., Buerki, S., Schmidt‐Trucksäss, A., & Mohaupt, M. G. (2022) · Life · doi:10.3390/life12111736 · Cited by 5 · see citing works
Adiponectin rescues synaptic plasticity in the dentate gyrus of a mouse model of Fragile X Syndrome
Thacker, J. S., Bettio, L. E. B., Liang, S., Shkolnikov, I., Collingridge, G. L., & Christie, B. R. (2024) · Philosophical Transactions of the Royal Society B Biological Sciences · doi:10.1098/rstb.2023.0221 · Cited by 4 · see citing works
FMR1 Disorders: Basics of Biology and Therapeutics in Development
Gillett, D. A., Tigro, H., Wang, Y., & Suo, Z. (2024) · Cells · doi:10.3390/cells13242100 · Cited by 4 · see citing works
Inequities in diagnosis of Fragile X syndrome in Colombia
Saldarriaga, W., Cabal‐Herrera, A. M., Fandiño‐Losada, A., Vásquez, A., Hagerman, R. J., & Tassone, F. (2021) · Journal of Applied Research in Intellectual Disabilities · doi:10.1111/jar.12863 · Cited by 4 · see citing works
Self-citationPharmacological Treatments for Fragile X Syndrome Based on Synaptic Dysfunction
Telias, M. (2019) · Current Pharmaceutical Design · doi:10.2174/1381612825666191102165206 · Cited by 4 · see citing works
Zhu, Y., Li, D., Hu, C., Tian, Y., Lu, P., Hagerman, R. J., Xu, X., & Xu, Q. (2025) · Molecular Autism · doi:10.1186/s13229-025-00691-z · Cited by 3 · see citing works
Self-citationMoreno, R. J., Rose, D. R., & Ashwood, P. (2026) · Journal of Neuroinflammation · doi:10.1186/s12974-026-03701-w · Cited by 3 · see citing works
Stanfield, A. C., & McKechanie, A. G. (2025) · Expert Review of Neurotherapeutics · doi:10.1080/14737175.2025.2510408 · Cited by 3 · see citing works
Molecular mechanisms for targeted treatments in fragile X syndrome
Miranda, I., & Hagerman, R. J. (2023) · Rare Disease and Orphan Drugs Journal · doi:10.20517/rdodj.2023.21 · Cited by 3 · see citing works
Self-citationEnomoto, Y., Shiromizu, T., Yasojima, S., Koiwa, J., Kuroda, Y., Ito, H., Yuge, M., Ohkawa, M., Shibata, R., Murakami, H., et al. (2025) · European Journal of Human Genetics · doi:10.1038/s41431-025-01815-y · Cited by 3 · see citing works
Do metabolic deficits contribute to sleep disruption in monogenic intellectual disability syndromes?
Valencia, M. L., Sofela, F. A., Jongens, T. A., & Sehgal, A. (2024) · Trends in Neurosciences · doi:10.1016/j.tins.2024.06.006 · Cited by 2 · see citing works
Medical use of cannabidiol in fragile X syndrome
Johnson, D., & Hagerman, R. J. (2023) · Elsevier eBooks · doi:10.1016/b978-0-323-90036-2.00029-6 · Cited by 1 · see citing works
Self-citationPrenatal Diagnosis and the Spectrum of Involvement from Fragile X Mutations
Hagerman, R. J., & Hagerman, P. J. (2021) · doi:10.1002/9781119676980.ch16
Self-citationFragile X-associated disorders
Hagerman, R. J. (2022) · Elsevier eBooks · doi:10.1016/b978-0-323-85654-6.00037-x
Self-citationSteenbergen, A. V., Minhas, A., Lin, T. R., Kaur, M., & Bolduc, F. V. (2023) · Research Square · doi:10.21203/rs.3.rs-3439158/v1
Systematic Review: Metformin, more than a hypoglycemic
Arce, J., & Sabattini, A. C. (2023) · SCT Proceedings in Interdisciplinary Insights and Innovations. · doi:10.56294/piii2023142
Chapter 65. Treatment of Individuals With Intellectual Developmental Disorder
Stratigos, K. A., Katz, J., & Hong, D. S. (2024) · American Psychiatric Association Publishing eBooks · doi:10.1176/appi.books.9781615375523.as65
Fragile X clinical features and neurobiology
Leigh, M. J., & Hagerman, R. J. (2024) · Elsevier eBooks · doi:10.1016/b978-0-443-19041-4.00036-4
Self-citationHasan, H., Santos, E. R., Amrei, S. A. M., Tassone, F., Randol, J. L., Hagerman, P. J., & Hagerman, R. J. (2025) · Case Reports in Genetics · doi:10.1155/crig/9751565
Self-citationSleep in a mouse model of fragile X syndrome is resistant to metabolic manipulations
Valencia, M. L., Aponte, R. A. V., Baur, J. A., Jongens, T. A., & Sehgal, A. (2025) · Human Molecular Genetics · doi:10.1093/hmg/ddaf149
The endocannabinoid system in intellectual disability
Vázquez-Oliver, A., Reyes‐Ramírez, L. D. L., Galera-López, L., & Ozaita, A. (2025) · Handbook of behavioral neuroscience · doi:10.1016/b978-0-443-13908-6.00022-0
Marsal-García, L., Choi, J.-H., Peraldi, E., Wu, P. Y., Dang, C. L., McKinney, R. A., Gantois, I., & Sonenberg, N. (2025) · Neurobiology of Disease · doi:10.1016/j.nbd.2025.107217 · Cited by 1 · see citing works
Abbeduto, L., Berry‐Kravis, E., Sterling, A. M., Sherman, S., Edgin, J. O., McDuffie, A. S., Hoffmann, A., Hamilton, D., Nelson, M., Aschkenasy, J. R., et al. (2020) · Figshare · doi:10.6084/m9.figshare.12023523
Protic, D. D., Stojković, M., & Budimirovic, D. B. (2025) · Medicinska istrazivanja · doi:10.5937/medi0-60213
BKCa channels as therapeutic targets in neurodevelopmental disorders : focus on acoustic dysfunction
Ferraguto, C. (2024) · theses.fr (ABES) · doi:10.70675/38217611z1b01z43f4zb4b0z72d28aca79c3
Gao, P., Wu, Y., Rumpf, S.-L., Weilin, C. H. E. N., Schaffer, P., Kolb, C., Giesbertz, P., Feng, X., Rodrigues, L. D., He, Y., et al. (2026) · Research Square · doi:10.21203/rs.3.rs-10296980/v1
Huang, W., Jiang, L., Zhu, Y., Duan, R., Xue, J., & Xia, Q. (2026) · Molecular Neurobiology · doi:10.1007/s12035-026-06115-0
Lippé, S., Buckser, R., Davoudi, S., Arutiunian, V., Knoth, I. S., Thurman, A., Bélanger, A.-M., Martin, C.-O., Proteau-Lemieux, M., Santos, E., et al. (2026) · Research Square · doi:10.21203/rs.3.rs-10924566/v1
Self-citation
- Authors
- Biag, H. M. B., Potter, L. A., Wilkins, V., Afzal, S., Rosvall, A., Salcedo‐Arellano, M. J., Rajaratnam, A., Manzano‐Nunez, R., Schneider, A., Tassone, F., Rivera, S. M., & Hagerman, R. J.
- Affiliations
- University of California Davis Medical Center (US)
- Primary Children's Hospital (US)
- Medical Investigation of Neurodevelopmental Disorders (MIND) Institute University of California Davis Medical Center Sacramento California
- Fundación Valle del Lili Clinical Research Center Cali Colombia
- Corresponding author
- Biag, Hazel Maridith B.
- ORCID
- Venue
- Molecular Genetics & Genomic Medicine
- Publisher
- Wiley
- Published
- 2019-09-14
- Volume
- 7
- Issue
- 11
- Pages
- e956-e956
- ISSN
- 2324-9269 (ISSN-L)
- PMC
- PMC6825840
- OpenAlex
- W2972397981
- Cited by
- 68
- References
- 49
- Citations by year
- 2026: 4 · 2025: 10 · 2024: 17 · 2023: 9 · 2022: 11 · 2021: 7 · 2020: 9 · 2019: 1
- FWCI
- 4.20
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Licence
- Locations
- Molecular Genetics & Genomic Medicine (publishedVersion, cc-by-nc-nd)
- PubMed Central (publishedVersion, cc-by-nc-nd)
- PubMed (publishedVersion)
- eScholarship (California Digital Library) (submittedVersion, cc-by-nc-nd)
- DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion)
- eScholarship (California Digital Library) (submittedVersion)
- Funders
- Intellectual and Developmental Disabilities Research Center (U54 HD079125)
- Azrieli Foundation
- National Institutes of Health (UL1 TR001860)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Life Sciences › Neuroscience › Cognitive Neuroscience › Autism Spectrum Disorder Research
- Life Sciences › Neuroscience › Developmental Neuroscience › Williams Syndrome Research
- Keywords
- metformin, fragile X syndrome, young children, behavioral symptoms, language development, lethargy, stereotypy
- MeSH terms
- Child, Child, Preschool, Fragile X Syndrome, Humans, Hypoglycemic Agents, Male, Metformin, Prognosis, Neurodevelopmental Disorders
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