A controlled trial of stimulant medication in children with the fragile X syndrome
Abstract
Attentional deficits and hyperactivity frequently are major problems for fra (X) boys. This study evaluated the effectiveness of 2 stimulant medications, methylphenidate and dextroamphetamine compared to placebo in 15 children (13 males, 2 females) with the fra (X) syndrome. A double‐blind crossover design was used with outcome measures which included parent and teacher behavior checklists, a controlled observation period, continuous performance tasks and an actometer measure of movement. When the children were treated with methylphenidate only, improvement was seen in socialization skills and attention span according to teacher checklists. Ten children were clinically considered responders and treatment was continued after the study was completed.
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Initial randomized controlled trial data of response to methylphenidate and dextroamphetamine suggest moderate response rates on attention and social skills in FXS.
“with initial randomized controlled trial data of response to methylphenidate and dextroamphetamine suggesting moderate response rates on attention and social skills [731].”
From Reward circuitry dysfunction in psychiatric and neurodevelopmental disorders and genetic syndromes: animal models and clinical findings (2012) · cites this paper as [731]
Cites
Kaufman Assessment Battery for Children
Kaufman, A. S., & Kaufman, N. L. (1983) · PsycTESTS Dataset · doi:10.1037/t27677-000 · Cited by 1,011 · see citing works
Handbook of Mental Deficiency: Psychological Theory and Research
Ellis, N. R. (2018) · Medical Entomology and Zoology · Cited by 635 · see citing works
Rating scales for use in drug studies with children
Conners, C. K. (1973) · Cited by 589 · see citing works
A REVIEW OF STIMULANT DRUG RESEARCH WITH HYPERACTIVE CHILDREN
Barkley, R. A. (1977) · Journal of Child Psychology and Psychiatry · doi:10.1111/j.1469-7610.1977.tb00425.x · Cited by 542 · see citing works
Sprague, R. L., & Sleator, E. K. (1977) · Science · doi:10.1126/science.337493 · Cited by 441 · see citing works
Hyperactive Children: A Handbook for Diagnosis and Treatment
Barkley, R. A. (1981) · Medical Entomology and Zoology · Cited by 408 · see citing works
Hyperactive Children: A Handbook for Diagnosis and Treatment
Wender, E. H. (1982) · Archives of Pediatrics and Adolescent Medicine · doi:10.1001/archpedi.1982.03970430087038 · Cited by 386 · see citing works
Conners' Teacher Rating Scale for use in drug studies with children ? An empirical study
WERRY, J. S., Sprague, R. L., & Cohen, M. N. (1976) · Research on Child and Adolescent Psychopathology · doi:10.1007/bf00916752 · Cited by 268 · see citing works
Foster, N. (2011) · Springer eBooks · doi:10.1007/978-0-387-79061-9_298 · Cited by 194 · see citing works
BENZEDRINE® AND DEXEDRINE® IN THE TREATMENT OF CHILDREN'S BEHAVIOR DISORDERS
Bradley, C. (1950) · PEDIATRICS · doi:10.1542/peds.5.1.24 · Cited by 157 · see citing works
Barkley, R. A., Fischer, M., Newby, R. F., & Breen, M. J. (1988) · Journal of Clinical Child Psychology · doi:10.1207/s15374424jccp1701_3 · Cited by 149 · see citing works
Chudley, A. E., & Hagerman, R. J. (1987) · The Journal of Pediatrics · doi:10.1016/s0022-3476(87)80392-x · Cited by 148 · see citing works
Self-citationEffects of Psychopharmacologic Agents on Learning Disorders
Sprague, R. L., & Sleator, E. K. (1973) · Pediatric Clinics of North America · doi:10.1016/s0031-3955(16)32892-9 · Cited by 138 · see citing works
The psychological profile of the fragile X syndrome
Fryns, J. P., Jacobs, J., Kleczkowska, A., & BERGHE, H. V. D. (1984) · Clinical Genetics · doi:10.1111/j.1399-0004.1984.tb00474.x · Cited by 121 · see citing works
The assessment of impulsivity and mediating behaviors in hyperactive and nonhyperactive boys
Gordon, M. (1979) · Research on Child and Adolescent Psychopathology · doi:10.1007/bf00916541 · Cited by 115 · see citing works
A new rating scale for diagnosis and monitoring of ADD children.
Rk, U., Ek, S., & Rl, S. (1984) · PubMed · Cited by 102 · see citing works
The fragile X syndrome. Diagnosis, biochemistry, and intervention.
Hecht, F. M., & Hecht, B. K. (1984) · Europe PMC (PubMed Central) · Cited by 101 · see citing works
AN OBJECTIVE MEASURE OF PHYSICAL ACTIVITY FOR EPIDEMIOLOGIC RESEARCH
LaPorte, R. E., Kuller, L. H., Kupfer, D. J., McPartland, R. J., Matthews, G. A., & Caspersen, C. J. (1979) · American Journal of Epidemiology · doi:10.1093/oxfordjournals.aje.a112671 · Cited by 101 · see citing works
McClure, F. D., & Gordon, M. (1984) · Research on Child and Adolescent Psychopathology · doi:10.1007/bf00916850 · Cited by 93 · see citing works
Aman, M. G. (1982) · Journal of Autism and Developmental Disorders · doi:10.1007/bf01538326 · Cited by 84 · see citing works
Learning Disabilities and Attentional Problems in Boys With the Fragile X Syndrome
Hagerman, R. J. (1985) · Archives of Pediatrics and Adolescent Medicine · doi:10.1001/archpedi.1985.02140090036021 · Cited by 82 · see citing works
Self-citationDevelopmental and behavioural disturbances in 13 boys with fragile X syndrome
Largo, R. H., & Schinzel, A. (1985) · European Journal of Pediatrics · doi:10.1007/bf00442299 · Cited by 80 · see citing works
Natural social behaviors in hyperactive children: Dose effects of methylphenidate.
Whalen, C. K., Henker, B. A., Swanson, J. M., Granger, D. A., Kliewer, W. L., & Spencer, J. (1987) · Journal of Consulting and Clinical Psychology · doi:10.1037/0022-006x.55.2.187 · Cited by 71 · see citing works
IS THE FRAGILE X SYNDROME AMENABLE TO TREATMENT?
LEJEUNE, J. (1982) · The Lancet · doi:10.1016/s0140-6736(82)90991-6 · Cited by 70 · see citing works
Oral folic acid versus placebo in the treatment of males with the fragile X syndrome
Hagerman, R. J., Jackson, A. W., Levitas, A. S., Braden, M. L., McBogg, P., Kemper, M. B., McGavran, L., Berry, R. M., Matus, I., Hagerman, P. J., et al. (1986) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320230119 · Cited by 65 · see citing works
Self-citationNeurological findings in patients with the fragile-X syndrome.
Finelli, P. F., Pueschel, S. M., Padre-Mendoza, T., & O'Brien, M. M. (1985) · Journal of Neurology Neurosurgery & Psychiatry · doi:10.1136/jnnp.48.2.150 · Cited by 53 · see citing works
Methylphenidate in severely retarded residents and the clinical significance of stereotypic behavior
Aman, M. G., & Singh, N. N. (1982) · Applied Research in Mental Retardation · doi:10.1016/s0270-3092(82)80002-7 · Cited by 47 · see citing works
Prevalence and pattern of drug treatment for behavior and seizure disorders of TMR students.
Gadow, K. D., & Kalachnik, J. E. (1981) · PubMed · Cited by 43 · see citing works
Responders, Nonresponders, and Placebo Responders Among Children with Attention Deficit Disorder
Ullmann, R. K., & Sleator, E. K. (1986) · Clinical Pediatrics · doi:10.1177/000992288602501201 · Cited by 42 · see citing works
Alexandris, A., & Lundell, F. W. (1968) · PubMed · Cited by 40 · see citing works
Folic acid therapy in the fragile X syndrome
Brown, W. T., Jenkins, E., Friedman, E., et al. (1984) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320170121 · Cited by 40 · see citing works
Attention Deficit Disorder Children with or without Hyperactivity
Ullmann, R. K., & Sleator, E. K. (1985) · Clinical Pediatrics · doi:10.1177/000992288502401001 · Cited by 37 · see citing works
Folic acid treatment in males and females with fragile‐(X)‐syndrome
Froster‐Iskenius, U., Bödeker, K., Oepen, T., Matthes, R., Piper, U., SCHWINGER, E., Opitz, J. M., & Reynolds, J. F. (1986) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320230121 · Cited by 36 · see citing works
Folic acid therapy in the fragile X syndrome
Erbe, R. W., & Opitz, J. M. (1984) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320170122 · Cited by 35 · see citing works
High dose folic acid treatment of fragile (X) males
Brown, W. T., Cohen, I. L., Fisch, G. S., et al. (1986) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320230120 · Cited by 31 · see citing works
Effect of folic acid treatment in the fragile X syndrome
Gustavson, K., Dahlbom, K., Flood, A., et al. (1985) · Clinical Genetics · doi:10.1111/j.1399-0004.1985.tb00232.x · Cited by 26 · see citing works
Blacklidge, V. Y., & Ekblad, R. L. (1971) · PEDIATRICS · doi:10.1542/peds.47.5.923 · Cited by 21 · see citing works
A Controlled Study of Folic Acid Treatment in Three Fragile X Syndrome Males
Madison, L. S., Wells, T. E., FRISTO, T. E., & Benesch, C. G. (1986) · Journal of Developmental & Behavioral Pediatrics · doi:10.1097/00004703-198608000-00007 · Cited by 13 · see citing works
Medical genetics: past, present and future.
Abuelo, D. (1999) · PubMed · Cited by 13 · see citing works
Blood Level Measurement of Psychoactive Drugs in Pediatric Psychiatry
Gualtieri, C. T., Golden, R., Evans, R. W., et al. (1984) · Therapeutic Drug Monitoring · doi:10.1097/00007691-198406000-00001 · Cited by 11 · see citing works
Lacassie, Y., Curotto, B., Ma, A., et al. (1984) · PubMed · Cited by 5 · see citing works
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Hagerman, R. J., Berry-Kravis, E., Hazlett, H. C., Bailey, D. B., Moine, H., Kooy, R. F., Tassone, F., Gantois, I., Sonenberg, N., Mandel, J. L., et al. (2017) · Nature Reviews Disease Primers · doi:10.1038/nrdp.2017.65 · Cited by 838 · see citing works
Self-citationDrug development for neurodevelopmental disorders: lessons learned from fragile X syndrome
Berry-Kravis, E. M., Lindemann, L., Jønch, A. E., Apostol, G., Bear, M. F., Carpenter, R. L., Crawley, J. N., Curie, A., Des Portes, V., Hossain, F., et al. (2018) · Nature Reviews Drug Discovery · doi:10.1038/nrd.2017.221 · Cited by 336 · see citing works
Self-citationFARZIN, F., PERRY, H., HESSL, D., LOESCH, D., COHEN, J., BACALMAN, S., GANE, L., TASSONE, F., HAGERMAN, P., & HAGERMAN, R. (2006) · Journal of Developmental & Behavioral Pediatrics · doi:10.1097/00004703-200604002-00012 · Cited by 325 · see citing works
Self-citationDichter, G. S., Damiano, C. A., & Allen, J. A. (2012) · Journal of Neurodevelopmental Disorders · doi:10.1186/1866-1955-4-19 · Cited by 299 · see citing works
ADHD symptoms in children with FXS
Sullivan, K., Hatton, D., Hammer, J., Sideris, J., Hooper, S., Ornstein, P., & Bailey, D. (2006) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.31388 · Cited by 174 · see citing works
Psychopharmacology in fragile X syndrome—Present and future
Berry‐Kravis, E., & Potanos, K. (2004) · Mental Retardation and Developmental Disabilities Research Reviews · doi:10.1002/mrdd.20007 · Cited by 151 · see citing works
Torrioli, M. G., Vernacotola, S., Peruzzi, L., Tabolacci, E., Mila, M., Militerni, R., Musumeci, S., Ramos, F. J., Frontera, M., Sorge, G., et al. (2008) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.32268 · Cited by 100 · see citing works
FMR1 premutation and full mutation molecular mechanisms related to autism
Hagerman, R., Au, J., & Hagerman, P. (2011) · Journal of Neurodevelopmental Disorders · doi:10.1007/s11689-011-9084-5 · Cited by 85 · see citing works
Self-citationTreatment with valproic acid ameliorates ADHD symptoms in fragile X syndrome boys
Torrioli, M., Vernacotola, S., Setini, C., Bevilacqua, F., Martinelli, D., Snape, M., Hutchison, J. A., Di Raimo, F. R., Tabolacci, E., & Neri, G. (2010) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.33484 · Cited by 71 · see citing works
Screening for fragile X syndrome.
Murray, Cuckle, Taylor, & Hewison (1997) · Health Technology Assessment · doi:10.3310/hta1040 · Cited by 60 · see citing works
Treatments for fragile X syndrome: A closer look at the data
Hall, S. S. (2009) · Developmental Disabilities Research Reviews · doi:10.1002/ddrr.78 · Cited by 56 · see citing works
The use of actigraphy in the monitoring of methylphenidate versus placebo in ADHD: a meta-analysis
De Crescenzo, F., Armando, M., Mazzone, L., Ciliberto, M., Sciannamea, M., Figueroa, C., Janiri, L., Quested, D., & Vicari, S. (2014) · ADHD Attention Deficit and Hyperactivity Disorders · doi:10.1007/s12402-013-0122-x · Cited by 48 · see citing works
Turk, J. (2011) · Current Opinion in Psychiatry · doi:10.1097/yco.0b013e328349bb77 · Cited by 35 · see citing works
Understanding fragile X syndrome
Berry-Kravis, E., Grossman, A. W., Crnic, L. S., & Greenough, W. T. (2002) · Current Paediatrics · doi:10.1054/cupe.2002.0305 · Cited by 29 · see citing works
Blink rate in boys with fragile X syndrome: preliminary evidence for altered dopamine function
Roberts, J. E., Symons, F. J., Johnson, A. ‐., Hatton, D. D., & Boccia, M. L. (2005) · Journal of Intellectual Disability Research · doi:10.1111/j.1365-2788.2005.00713.x · Cited by 24 · see citing works
Results of a Nation-Wide Survey Evaluating Psychotropic Medication Use in Fragile X Syndrome
Valdovinos, M. G., Parsa, R. A., & Alexander, M. L. (2009) · Journal of Developmental and Physical Disabilities · doi:10.1007/s10882-008-9123-7 · Cited by 23 · see citing works
- Authors
- Hagerman, R. J., Murphy, M. A., & Wittenberger, M. D.
- Affiliations
- University of Colorado Health (US)
- Corresponding author
- Hagerman, Randi J.
- ORCID
- Hagerman, Randi J.
- Venue
- American Journal of Medical Genetics
- Publisher
- Wiley
- Published
- 1988-05
- Volume
- 30
- Issue
- 1-2
- Pages
- 377-392
- ISSN
- 0148-7299 (ISSN-L)
- 1096-8628
- OpenAlex
- W2136765120
- Cited by
- 158
- References
- 41
- Citations by year
- 2025: 2 · 2024: 7 · 2023: 1 · 2022: 2 · 2021: 1 · 2020: 3 · 2019: 2 · 2018: 3 · 2017: 7 · 2016: 3 · 2015: 3 · 2014: 4 · 2013: 8 · 2012: 4
- FWCI
- 2.66
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- American Journal of Medical Genetics (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
- Health Sciences › Medicine › Psychiatry and Mental health › Attention Deficit Hyperactivity Disorder
- Life Sciences › Neuroscience › Cognitive Neuroscience › Autism Spectrum Disorder Research
- Keywords
- fragile X syndrome, stimulant medication, methylphenidate, d-amphetamine, attention deficit hyperactivity disorder, attention span, socialization skills, children, treatment response
- MeSH terms
- Attention Deficit Disorder with Hyperactivity, Child, Child, Preschool, Clinical Trials as Topic, Dextroamphetamine, Double-Blind Method, Female, Fragile X Syndrome, Humans, Male, Methylphenidate, Sex Chromosome Aberrations
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