ADHD symptoms in children with FXS
Abstract
Parent‐ and teacher‐report of attention‐deficit/hyperactivity disorder (ADHD) symptoms were examined using problem behavior and DSM‐IV symptom inventory questionnaires for 63 children with full mutation fragile X syndrome (FXS) and 56 children without disabilities matched on mental age (MA). Prevalence rates of ADHD symptoms varied depending on type of measure (problem behavior or DSM‐IV criteria), subscale (ADHD‐inattentive or ADHD‐hyperactive), scoring method (continuous T‐scores or categorical scores based on DSM‐IV algorithm), and rater (parent or teacher). Overall, 54–59% of boys with FXS met diagnostic behavioral criteria for either ADHD‐inattentive type only, ADHD‐hyperactive type only, or ADHD‐combined type based on parent or teacher report. Boys with FXS were rated as having clinically high scores or met diagnostic criteria at higher rates than expected for the general population and had higher raw scores than their MA‐matched peers. Parent ratings of boys with FXS resulted in higher ADHD‐inattentive type and ADHD‐hyperactive type T‐scores than teachers. Boys who were rated as meeting DSM‐IV criteria were more likely to be taking psychotropic medication and to have younger mental ages. Parents were substantially more likely than teachers to rate boys as meeting DSM‐IV criteria for ADHD‐inattentive type, while teachers were only slightly more likely than parents to rate boys as meeting DSM‐IV criteria for ADHD‐hyperactive type. Teachers were more likely than parents to rate boys as meeting DSM‐IV criteria for ADHD when boys had lower levels of FMRP. © 2006 Wiley‐Liss, Inc.
Integrity signals
Registry
Cites
Statistics
Claims the paper makes
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
Boys with fragile X syndrome typically show ADHD with impulsivity and anxiety, repetitive language, hand biting, hand stereotypies, rocking, and sometimes head banging.
“The behavior of boys with FXS typically includes attention deficit hyperactivity disorder (ADHD), with significant impulsivity and anxiety, as well as behaviors that include repetitive language, hand biting, hand stereotypies, rocking, and sometimes head banging (24,25).”
From Fragile X syndrome: causes, diagnosis, mechanisms, and therapeutics (2012) · cites this paper as [25]
Cites
Diagnostic and Statistical Manual of Mental Disorders
Williams, J. B. W., & First, M. B. (2013) · Oxford University Press eBooks · doi:10.1093/acrefore/9780199975839.013.104 · Cited by 109,841 · see citing works
Controlling the False Discovery Rate: A Practical and Powerful Approach to Multiple Testing
Benjamini, Y., & Hochberg, Y. (1995) · Journal of the Royal Statistical Society Series B: Statistical Methodology · doi:10.1111/j.2517-6161.1995.tb02031.x · Cited by 109,482 · see citing works
Hierarchical Linear Models: Applications and Data Analysis Methods.
Longford, N. T., Bryk, A. S., & Raudenbush, S. W. (1993) · Contemporary Sociology A Journal of Reviews · doi:10.2307/2075823 · Cited by 18,768 · see citing works
Hierarchical Linear Models: Applications and Data Analysis Methods
Davidian, M. (2003) · Journal of the American Statistical Association · doi:10.1198/jasa.2003.s288 · Cited by 13,820 · see citing works
Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, Text Revision (DSM-IV-TR)
American Psychiatric Association (2000) · doi:10.1176/appi.books.9780890423349 · Cited by 11,069 · see citing works
(1991) · Medical Entomology and Zoology · Cited by 8,071 · see citing works
Manual for the Teacher's Report Form and 1991 Profile
Achenbach, T. M. (1991) · Medical Entomology and Zoology · Cited by 2,042 · see citing works
Committee on Quality Improvement, S. O. A.-D. D. (2000) · Pediatrics · doi:10.1542/peds.105.5.1158 · Cited by 1,076 · see citing works
Leiter International Performance Scale-Revised
Sullivan, S. A. (1998) · Psychology in the Schools · doi:10.1002/(sici)1520-6807(199804)35:2 · Cited by 1,012 · see citing works
Mapping of DNA instability at the fragile X to a trinucleotide repeat sequence p(CCG)n
Kremer, E., Pritchard, M., Lynch, M., Yu, S., Holman, K., Baker, E., Warren, S., Schlessinger, D., Sutherland, G., & Richards, R. (1991) · Science · doi:10.1126/science.1675488 · Cited by 929 · see citing works
FMR1 and the fragile X syndrome: Human genome epidemiology review
Crawford, D. C., Acuña, J. M., & Sherman, S. L. (2001) · Genetics in Medicine · doi:10.1097/00125817-200109000-00006 · Cited by 711 · see citing works
Weiler, I. J., Irwin, S. A., Klintsova, A. Y., Spencer, C. M., Brazelton, A. D., Miyashiro, K., Comery, T. A., Patel, B., Eberwine, J., & Greenough, W. T. (1997) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.94.10.5395 · Cited by 644 · cited 2× · see citing works
“Perhaps because FMRP has been found to be involved in synaptic functioning [Weiler et al., 1997], as well as theorized to alter structures in the brain [Reiss et al., 1995; Eliez et al., 2001; Hessl et al., 2004], FXS probably affects cognitive function, including attention, in an ongoing dynamic…”
BackgroundRNA and microRNAs in fragile X mental retardation
Jin, P., Alisch, R. S., & Warren, S. T. (2004) · Nature Cell Biology · doi:10.1038/ncb1104-1048 · Cited by 344 · see citing works
Dombrowski, C. (2002) · Human Molecular Genetics · doi:10.1093/hmg/11.4.371 · Cited by 289 · see citing works
Specification of the Neurobehavioral Phenotype in Males with Fragile X Syndrome
Baumgardner, T. L., Reiss, A. L., Freund, L. S., & Abrams, M. T. (1995) · PEDIATRICS · doi:10.1542/peds.95.5.744 · Cited by 245 · see citing works
Requirement of GTP Hydrolysis for Dissociation of the Signal Recognition Particle from Its Receptor
Connolly, T., Rapiejko, P. J., & Gilmore, R. (1991) · Science · doi:10.1126/science.252.5009.1171 · Cited by 223 · see citing works
Visual search in typically developing toddlers and toddlers with Fragile X or Williams syndrome.
Scerif, G., Cornish, K., Wilding, J., Driver, J., & Karmiloff-Smith, A. (2004) · Developmental Science · doi:10.1111/j.1467-7687.2004.00327.x · Cited by 202 · see citing works
Psychiatric Disorders Associated With Fragile X in the Young Female
Freund, L. S., Reiss, A. L., & Abrams, M. T. (1993) · PEDIATRICS · doi:10.1542/peds.91.2.321 · Cited by 199 · see citing works
A neuropsychological profile of attention deficits in young males with fragile X syndrome
Munir, F., Cornish, K. M., & Wilding, J. P. H. (2000) · Neuropsychologia · doi:10.1016/s0028-3932(00)00036-1 · Cited by 188 · see citing works
Estimating Individual Developmental Functions: Methods and Their Assumptions
Burchinal, M., & Appelbaum, M. I. (1991) · Child Development · doi:10.1111/j.1467-8624.1991.tb01512.x · Cited by 183 · see citing works
Rapid antibody test for fragile X syndrome
Willemsen, R., Mohkamsing, S., de Vries, B., van den Ouweland, A., GALJAARD, H., Oostra, B. A., Devys, D., & Mandel, J. (1995) · The Lancet · doi:10.1016/s0140-6736(95)90979-6 · Cited by 178 · see citing works
Attention, 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
Brain anatomy, gender and IQ in children and adolescents with fragile X syndrome
D, S. E. M. (2001) · Brain · doi:10.1093/brain/124.8.1610 · Cited by 169 · see citing works
The neuroanatomy and neuroendocrinology of fragile X syndrome
Hessl, D., Rivera, S. M., & Reiss, A. L. (2004) · Mental Retardation and Developmental Disabilities Research Reviews · doi:10.1002/mrdd.20004 · Cited by 165 · see citing works
A controlled trial of stimulant medication in children with the fragile X syndrome
Hagerman, R. J., Murphy, M. A., & Wittenberger, M. D. (1988) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320300138 · Cited by 158 · 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
Fragile X syndrome: genetic predisposition to psychopathology
Bregman, J. D., Leckman, J. F., & Ort, S. I. (1988) · Journal of Autism and Developmental Disorders · doi:10.1007/bf02212191 · Cited by 150 · see citing works
Problem behavior in boys with fragile X syndrome
Hatton, D. D., Hooper, S. R., Bailey, D. B., Skinner, M. L., Sullivan, K. M., & Wheeler, A. (2002) · American Journal of Medical Genetics · doi:10.1002/ajmg.10216 · Cited by 149 · see citing works
Self-citationSocial behavior profile in young males with fragile X syndrome: Characteristics and specificity
Kau, A. S. M., Tierney, E., Bukelis, I., Stump, M. H., Kates, W. R., Trescher, W. H., & Kaufmann, W. E. (2004) · American Journal of Medical Genetics Part A · doi:10.1002/ajmg.a.20218 · Cited by 149 · see citing works
Attention and language in fragile X
Cornish, K. M., Sudhalter, V., & Turk, J. (2004) · Mental Retardation and Developmental Disabilities Research Reviews · doi:10.1002/mrdd.20003 · Cited by 136 · see citing works
Intelligence and cognitive function in children and adolescents with spinal muscular atrophy
von Gontard, A., Zerres, K., Backes, M., et al. (2002) · Neuromuscular Disorders · doi:10.1016/s0960-8966(01)00274-7 · Cited by 129 · see citing works
Further delineation of the executive deficit in males with fragile-X syndrome
Wilding, J. P. H., Cornish, K. M., & Munir, F. (2002) · Neuropsychologia · doi:10.1016/s0028-3932(01)00212-3 · Cited by 128 · see citing works
Role of Religion in the Lives of Latino Families of Young Children With Developmental Delays
Skinner, D., Correa, V. I., Skinner, M. L., & Bailey, D. B. (2001) · American Journal on Mental Retardation · doi:10.1352/0895-8017(2001)106 · Cited by 124 · see citing works
Self-citationThe 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
Cornish, K. M., Munir, F., & Cross, G. S. (2001) · Journal of Cognitive Neuroscience · doi:10.1162/089892901564126 · Cited by 118 · see citing works
Hagerman, R. J., & Hagerman, P. J. (2002) · Cambridge University Press eBooks · doi:10.1017/cbo9780511543876.009 · Cited by 107 · see citing works
Cai, X., Kaiser, A. P., & Hancock, T. B. (2004) · Journal of Clinical Child & Adolescent Psychology · doi:10.1207/s15374424jccp3302_12 · Cited by 103 · see citing works
Pearson, D. A., Santos, C. W., Roache, J. D., Casat, C. D., Loveland, K. A., Lachar, D., LANE, D. M., Faria, L. P., & Cleveland, L. A. (2003) · Journal of the American Academy of Child & Adolescent Psychiatry · doi:10.1097/00004583-200302000-00015 · Cited by 96 · see citing works
Fragile (X) syndrome: a study of the psychological profile in 23 prepubertal patients
Borghgraef, M., Fryns, J. P., Dlelkens, A., Pyck, K., & BERGHE, H. V. D. (1987) · Clinical Genetics · doi:10.1111/j.1399-0004.1987.tb03351.x · Cited by 93 · see citing works
Stability of Behavioral Ratings of Children with SLI
Redmond, S. M., & Rice, M. L. (2002) · Journal of Speech Language and Hearing Research · doi:10.1044/1092-4388(2002/014 · Cited by 91 · see citing works
Social Functioning Among Girls with Fragile X or Turner Syndrome and Their Sisters
Mazzocco, M. M. M., Baumgardner, T. L., Freund, L. S., & Reiss, A. L. (1998) · Journal of Autism and Developmental Disorders · doi:10.1023/a:1026000111467 · Cited by 84 · see citing works
Hagerman, R. J. (1987) · Current Problems in Pediatrics · doi:10.1016/0045-9380(87)90011-9 · Cited by 80 · see citing works
Fragile X Syndrome and Attentional Deficits
Turk, J. (1998) · Journal of Applied Research in Intellectual Disabilities · doi:10.1111/j.1468-3148.1998.tb00060.x · Cited by 72 · see citing works
Long-Term Follow-Up of Children with Mental Retardation/Borderline Intellectual Functioning and ADHD
Handen, B., Janosky, J., & McAuliffe, S. (1997) · Journal of Abnormal Child Psychology · doi:10.1023/a:1025760302598 · Cited by 62 · see citing works
Abnormal behaviors of young girls with fragile X syndrome
Lachiewicz, A. M. (1992) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320430111 · Cited by 57 · see citing works
Behavioural and emotional disturbance in fragile X syndrome
Einfeld, S., Tonge, B. J., & Florio, T. (1994) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320510417 · Cited by 48 · see citing works
Pearson, D. A., Lane, D. M., Santos, C. W., Casat, C. D., Jerger, S. W., Loveland, K. A., Faria, L. P., Mansour, R., Henderson, J. A., Payne, C. D., et al. (2004) · Journal of the American Academy of Child & Adolescent Psychiatry · doi:10.1097/01.chi.0000120024.14101.96 · Cited by 45 · see citing works
A DSM-IV—Referenced Teacher Rating Scale for Use in Clinical Management
Mattison, R. E., Gadow, K. D., Sprafkin, J. N., Nolan, E. E., & Schneider, J. (2003) · Journal of the American Academy of Child & Adolescent Psychiatry · doi:10.1097/01.chi.0000046813.95464.b3 · Cited by 43 · see citing works
Lachiewicz, A. M., & Dawson, D. V. (1994) · American Journal of Medical Genetics · doi:10.1002/ajmg.1320510413 · Cited by 43 · see citing works
Mirrett, P., Roberts, J. E., & Price, J. R. (2003) · Language Speech and Hearing Services in Schools · doi:10.1044/0161-1461(2003/026 · Cited by 38 · see citing works
Never again joy without sorrow: The effect on parents of a child with ataxia-telangiectasia
Fanos, J. H., & Mackintosh, M. (1999) · American Journal of Medical Genetics · doi:10.1002/(sici)1096-8628(19991222)87:5 · Cited by 35 · see citing works
Fragile X premutations and (TA)n estrogen receptor polymorphism in women with ovarian dysfunction
Syrrou, M., Georgiou, I. A., Patsalis, P. C., Bouba, I., Adonakis, G., & Pagoulatos, G. N. (1999) · American Journal of Medical Genetics · doi:10.1002/(sici)1096-8628(19990528)84:3 · Cited by 33 · see citing works
Correspondence of parent and teacher reports in medication trials
Faraone, S. V., Biederman, J., & Zimmerman, B. A. (2005) · European Child & Adolescent Psychiatry · doi:10.1007/s00787-005-0415-5 · Cited by 32 · see citing works
Medication use in fragile X syndrome
Amaria, R. N., Billeisen, L. L., & Hagerman, R. J. (2001) · Cited by 27 · see citing works
Lobar holoprosencephaly in an infant born to a mother with classic phenylketonuria
Keller, K. M., McCune, H., Williams, C. A., & Muenke, M. (2000) · American Journal of Medical Genetics · doi:10.1002/1096-8628(20001113)95:2 · Cited by 12 · see citing works
Kallmann syndrome gene (KAL-X) is not mutated in schizophrenia
O’Neill, M. J., Brewer, W. J., Thornley, C., Copolov, D., Warne, G. L., Sinclair, A. H., Forrest, S., & Williamson, R. (1999) · American Journal of Medical Genetics · doi:10.1002/(sici)1096-8628(19990205)88:1 · Cited by 7 · see citing works
Personal Control and the Ecology of Community Living Settings: Beyond Living-Unit Size and Type
Stancliffe, R. J., Abery, B. H., & Smith, J. (2000) · American Journal on Mental Retardation · doi:10.1352/0895-8017(2000)105
Orsmond, G. I., Seltzer, M. M., Krauss, M. W., & Hong, J. (2003) · American Journal on Mental Retardation · doi:10.1352/0895-8017(2003)108
Flagged first, then by citations
Cited by
Sorted by citations
Elia, J., Gai, X., Xie, H. M., Perin, J. C., Geiger, E., Glessner, J. T., D'arcy, M., deBerardinis, R., Frackelton, E., Kim, C., et al. (2010) · Molecular Psychiatry · doi:10.1038/mp.2009.57 · Cited by 529 · see citing works
Fragile X syndrome: causes, diagnosis, mechanisms, and therapeutics
Bagni, C., Tassone, F., Neri, G., & Hagerman, R. (2012) · Journal of Clinical Investigation · doi:10.1172/jci63141 · Cited by 321 · see citing works
Autism spectrum disorder in fragile X syndrome: cooccurring conditions and current treatment
Kaufmann, W. E., Kidd, S. A., Andrews, H. F., Budimirovic, D. B., Esler, A., Haas-Givler, B., Stackhouse, T., Riley, C., Peacock, G., Sherman, S. L., et al. (2017) · PEDIATRICS · doi:10.1542/peds.2016-1159f · Cited by 268 · see citing works
Modeling fragile X syndrome in the <i>Fmr1</i> knockout mouse
Kazdoba, T. M., Leach, P. T., Silverman, J. L., & Crawley, J. N. (2014) · Intractable & Rare Diseases Research · doi:10.5582/irdr.2014.01024 · Cited by 241 · see citing works
FMRP Acts as a Key Messenger for Dopamine Modulation in the Forebrain
Wang, H., Wu, L.-J., Kim, S. S., Lee, F. J. S., Gong, B., Toyoda, H., Ren, M., Shang, Y.-Z., Xu, H., Liu, F., et al. (2008) · Neuron · doi:10.1016/j.neuron.2008.06.027 · Cited by 186 · see citing works
Fragile X syndrome: a review of clinical and molecular diagnoses
Ciaccio, C., Fontana, L., Milani, D., Tabano, S., Miozzo, M., & Esposito, S. (2017) · Italian Journal of Pediatrics · doi:10.1186/s13052-017-0355-y · Cited by 160 · see citing works
Social behavior in Fmr1 knockout mice carrying a human FMR1 transgene.
Spencer, C. M., Graham, D. F., Yuva-Paylor, L. A., Nelson, D. L., & Paylor, R. (2008) · Behavioral Neuroscience · doi:10.1037/0735-7044.122.3.710 · Cited by 91 · 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
Gene, brain, and behavior relationships in fragile X syndrome: Evidence from neuroimaging studies
Lightbody, A. A., & Reiss, A. L. (2009) · Developmental Disabilities Research Reviews · doi:10.1002/ddrr.77 · Cited by 85 · see citing works
Scerif, G., Longhi, E., Cole, V., Karmiloff‐Smith, A., & Cornish, K. (2012) · Journal of Child Psychology and Psychiatry · doi:10.1111/j.1469-7610.2011.02515.x · Cited by 65 · see citing works
Cornish, K., Cole, V., Longhi, E., Karmiloff-Smith, A., & Scerif, G. (2012) · American Journal on Intellectual and Developmental Disabilities · doi:10.1352/1944-7558-117.2.103 · Cited by 58 · see citing works
Visual attention and autistic behavior in infants with fragile X syndrome
Roberts, J. E., Hatton, D. D., Long, A. C. J., Anello, V., & Colombo, J. (2012) · Journal of Autism and Developmental Disorders · doi:10.1007/s10803-011-1316-8 · Cited by 50 · see citing works
Self-citationBehavioral Intervention for Problem Behavior in Children With Fragile X Syndrome
Moskowitz, L. J., Carr, E. G., & Durand, V. M. (2011) · American Journal on Intellectual and Developmental Disabilities · doi:10.1352/1944-7558-116.6.457 · Cited by 35 · see citing works
Astrocytes in fragile X syndrome
Talvio, K., & Castrén, M. L. (2024) · Frontiers in Cellular Neuroscience · doi:10.3389/fncel.2023.1322541 · Cited by 8 · see citing works
- Authors
- Sullivan, K., Hatton, D., Hammer, J., Sideris, J., Hooper, S., Ornstein, P., & Bailey, D.
- Affiliations
- University of North Carolina at Chapel Hill (US)
- Corresponding author
- Hatton, Deborah
- Venue
- American Journal of Medical Genetics Part A
- Publisher
- Wiley
- Published
- 2006-10-04
- Volume
- 140A
- Issue
- 21
- Pages
- 2275-2288
- ISSN
- 1552-4825 (ISSN-L)
- 1552-4833
- OpenAlex
- W2035631323
- Cited by
- 174
- References
- 59
- Citations by year
- 2026: 2 · 2025: 7 · 2024: 7 · 2023: 8 · 2022: 13 · 2021: 7 · 2020: 8 · 2019: 13 · 2018: 9 · 2017: 10 · 2016: 9 · 2015: 6 · 2014: 12 · 2013: 9 · 2012: 8
- FWCI
- 4.35
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- American Journal of Medical Genetics Part A (publishedVersion)
- PubMed (publishedVersion)
- Funders
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (1-R01-HD40602-01, R01HD040602)
- 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
- attention deficit hyperactivity disorder, fragile X syndrome, full mutation, inattentive subtype, combined type, parent report, teacher reports, rater differences, DSM-IV diagnostic criteria, FMRP, psychotropic drugs, behavioral problems, prevalence, boys
- MeSH terms
- Adolescent, Adolescent Behavior, Attention Deficit Disorder with Hyperactivity, Autistic Disorder, Biometry, Child, Child Behavior, Faculty, Fragile X Syndrome, Humans, Male, Parents, Surveys and Questionnaires, Case-Control Studies
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review