Metformin as targeted treatment in fragile X syndrome

Clinical Genetics

Volumn 93, Issue 2, 2018, Pages 216-222

  Dy, A B C ^a,b,c   Tassone, F ^c,e   Eldeeb, M ^c,e   Salcedo Arellano, M J ^c,e   Tartaglia, N ^d   Hagerman, R ^c,e  

^a ATENEO DE MANILA UNIVERSITY   (Philippines)

^b MedMom Institute for Human Development   (Philippines)

^c UNIVERSITY OF CALIFORNIA   (United States)

^d Children's Hospital Colorado   (United States)

^e UNIVERSITY OF CALIFORNIA   (United States)

Author keywords

fragile X syndrome; metformin; obesity; Prader Willi phenotype; targeted treatments

Indexed keywords

AMLODIPINE; AMPHETAMINE; ARIPIPRAZOLE; ATOMOXETINE; BASIMGLURANT; CALCIUM; CITALOPRAM; CYANOCOBALAMIN; DNA; ESCITALOPRAM; FLUVOXAMINE; GLUCOSE; GUANFACINE; HEMOGLOBIN A1C; LAMOTRIGINE; LITHIUM; LORATADINE; MAVOGLURANT; MEMANTINE; METFORMIN; METHYLPHENIDATE; MEVINOLIN; MINOCYCLINE; QUETIAPINE; RISPERIDONE; SERTRALINE; TRIAMTERENE; VALPROATE SEMISODIUM; VITAMIN D; ZIPRASIDONE; GLYCOSYLATED HEMOGLOBIN;

ABDOMINAL PAIN; ADOLESCENT; ADULT; AGGRESSION; AGITATION; AMNESIA; ANGER; ANXIETY; ARTICLE; ATTENTION DEFICIT DISORDER; AUTOMUTILATION; BEHAVIOR CHANGE; BINGE EATING DISORDER; BITE; BLOOD PRESSURE MEASUREMENT; BODY WEIGHT; BODY WEIGHT LOSS; CASE REPORT; CHILD; CLINICAL ARTICLE; COMPULSION; DELAYED PUBERTY; DIARRHEA; DIET RESTRICTION; DIVERGENT STRABISMUS; DRUG DOSE INCREASE; EATING HABIT; ENURESIS; EVENING DOSAGE; FEMALE; FOLLOW UP; FRAGILE X SYNDROME; GASTRIC BYPASS SURGERY; GASTROESOPHAGEAL REFLUX; HEADACHE; HEMOGLOBIN BLOOD LEVEL; HUMAN; HYPERACTIVITY; HYPERPHAGIA; HYPERTENSION; IMPAIRED GLUCOSE TOLERANCE; IMPULSIVENESS; INTELLIGENCE QUOTIENT; INTUBATION; IRRITABILITY; LOOSE FECES; LOW DRUG DOSE; MACROORCHIDISM; MALE; MEDICAL HISTORY; MIDDLE AGED; MORBID OBESITY; MORNING DOSAGE; MOSAICISM; MUSCLE HYPOTONIA; NON INSULIN DEPENDENT DIABETES MELLITUS; OBESITY; OVERNUTRITION; PHOBIA; PHYSICAL EXAMINATION; PRADER WILLI SYNDROME; PRESCHOOL CHILD; PRIORITY JOURNAL; PROBLEM BEHAVIOR; PROMINENT EAR; SCRATCHING; SELF ESTEEM; SENSORY DEFENSIVENESS; SOCIAL ADAPTATION; TREATMENT DURATION; VITAMIN BLOOD LEVEL; VITAMIN SUPPLEMENTATION; VOMITING; YOUNG ADULT; ANIMAL; BLOOD; GENETICS; GLUCOSE INTOLERANCE; MENTAL DISEASE; METABOLISM; PATHOLOGY; TREATMENT OUTCOME;

ADULT; ANIMALS; CHILD; CHILD, PRESCHOOL; DIABETES MELLITUS, TYPE 2; FEMALE; FRAGILE X SYNDROME; GLUCOSE INTOLERANCE; GLYCATED HEMOGLOBIN A; HUMANS; MALE; MENTAL DISORDERS; METFORMIN; MIDDLE AGED; OBESITY; PRADER-WILLI SYNDROME; TREATMENT OUTCOME; YOUNG ADULT;

EID: 85030655469     PISSN: 00099163     EISSN: 13990004     Source Type: Journal    
DOI: 10.1111/cge.13039     Document Type: Article

References (23)

1. Kidd SA, Lachiewicz A, Barbouth D, et al. Fragile X syndrome: a review of associated medical problems. Pediatrics. 2014;134(5):995-1005. https://doi.org/10.1542/peds.2013-4301.
2. Muzar Z, Lozano R, Kolevzon A, Hagerman RJ. The neurobiology of the Prader-Willi phenotype of fragile X syndrome. Intractable Rare Dis Res. 2016;5(4):255-261.
3. Bailey DB, Raspa M, Olmsted M, Holiday DB. Co-occurring conditions associated with FMR1 gene variations: findings from a national parent survey. Am J Med Genet A. 2008;146(16):2060-2069. https://doi.org/10.1002/ajmg.a.32439.
4. Cordeiro L, Ballinger E, Hagerman R, Hessl D. Clinical assessment of DSM-IV anxiety disorders in fragile X syndrome: prevalence and characterization. J Neurodev Disord. 2011;3(1):57-67. https://doi.org/10.1007/s11689-010-9067-y.
5. Hagerman RJ, Hagerman PJ. Fragile X Syndrome: Diagnosis, Treatment and Research. 3rd ed. Baltimore, Maryland: JHU Press; 2002.
6. McLennan Y, Polussa J, Tassone F, Hagerman R. Fragile x syndrome. Curr Genomics. 2011;12(3):216-224. https://doi.org/10.2174/138920211795677886.
7. Tounian P, Frelut ML, Parlier G, et al. Weight loss and changes in energy metabolism in massively obese adolescents. Int J Obes Relat Metab Disord. 1999;23(8):830-837.
8. Nowicki ST, Tassone F, Ono MY, et al. The Prader-Willi phenotype of fragile X syndrome. J Dev Behav Pediatr. 2007;28(2):133-138. https://doi.org/10.1097/01.DBP.0000267563.18952.c9.
9. Hagerman RJ, Berry-Kravis E, Kaufmann WE, et al. Advances in the treatment of fragile X syndrome. Pediatrics. 2009;123(1):378-390. https://doi.org/10.1542/peds.2008-0317.
10. Klein DJ, Cottingham EM, Sorter M, Barton B, Morrison JA. A randomized, double-blind, placebo-controlled trial of metformin treatment of weight gain associated with initiation of atypical antipsychotic therapy in children and adolescents. Am J Psychiatry. 2006;163(12):2072-2079. https://doi.org/10.1176/appi.ajp.163.12.2072.
11. Jones KL, Arslanian S, Peterokova VA, Park JS, Tomlinson MJ. Effect of metformin in pediatric patients with type 2 diabetes: a randomized controlled trial. Diabetes Care. 2002;25(1):89-94. https://doi.org/10.2337/diacare.25.1.89.
12. Park MH, Kinra S, Ward KJ, White B, Viner RM. Metformin for obesity in children and adolescents: a systematic review. Diabetes Care. 2009;32(9):1743-1745. https://doi.org/10.2337/dc09-0258.
13. Anagnostou E, Aman MG, Handen BL, et al. Metformin for treatment of overweight induced by atypical antipsychotic medication in young people with autism spectrum disorder. A randomized clinical trial. JAMA Psychiat. 2016;73(9):928-937. https://doi.org/10.1001/jamapsychiatry.2016.1232.
14. Ong CR, Molyneux LM, Constantino MI, Twigg SM. Long-term efficacy of metformin therapy in nonobese individuals with type 2 diabetes. Diabetes Care. 2006;29(11):2361-2364.
15. Bodmer M, Meier C, Krähenbühl S, Jick SS, Meier CR. Metformin, sulfonylureas, or other antidiabetes drugs and the risk of lactic acidosis or hypoglycemia a nested case–control analysis. Diabetes Care. 2008;31(11):2086-2091. https://doi.org/10.2337/dc08-1171.
16. Bolen S, Feldman L, Vassy J, et al. Systematic review: comparative effectiveness and safety of oral medications for type 2 diabetes mellitus. Ann Intern Med. 2007;147(6):386-399. https://doi.org/10.1001/archinte.147.6.386.
17. Barzilai N, Crandall JP, Kritchevsky SB, Espeland MA. Metformin as a tool to target aging. Cell Metab. 2016;23(6):1060-1065. https://doi.org/10.1016/j.cmet.2016.05.011.
18. Bannister CA, Holden SE, Jenkins-Jones S, et al. Can people with type 2 diabetes live longer than those without? A comparison of mortality in people initiated with metformin or sulphonylurea monotherapy and matched, non-diabetic controls. Diabetes Obes Metab. 2014;16(11):1165-1173. https://doi.org/10.1111/dom.12354.
19. Monyak RE, Emerson D, Schoenfeld BP, et al. Insulin Signaling Misregulation underlies Circadian and Cognitive Deficits in a Drosophila Fragile X Model. Molecular psychiatry. 2017;22(8):1140-1148. https://doi.org/10.1038/mp.2016.51.
20. Weisz ED, Monyak RE, Jongens TA. Deciphering discord: how Drosophila research has enhanced our understanding of the importance of FMRP in different spatial and temporal contexts. Exp Neurol. 2015;274:14-24. https://doi.org/10.1016/j.expneurol.2015.05.015.
21. Gantois I, Khoutorsky A, Popic J, et al. Metformin ameliorates core deficits in a mouse model of fragile X syndrome. Nat. Med. 2017;23(6):674-677. https://doi.org/10.1038/nm.4335.
22. Luo T, Nocon A, Fry J, et al. AMPK activation by metformin suppresses abnormal extracellular matrix remodeling in adipose tissue and ameliorates insulin resistance in obesity. Diabetes. 2016;65(8):2295-2310. https://doi.org/10.2337/db15-1122.
23. Greiss Hess L, Fitzpatrick SE, Nguyen DV, et al. A randomized, double-blind, placebo-controlled trial of low-dose sertraline in young children with fragile X syndrome. J Dev Behav Pediatr. 2016;37(8):619-628. https://doi.org/10.1097/DBP.0000000000000334.