ADHD symptoms in children with FXS

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.

[1]  D. Lane,et al.  Effects of methylphenidate treatment in children with mental retardation and ADHD: individual variation in medication response. , 2004, Journal of the American Academy of Child and Adolescent Psychiatry.

[2]  M. Appelbaum,et al.  Estimating Individual Developmental Functions: Methods and Their Assumptions , 1991 .

[3]  J. Wilding,et al.  A neuropsychological profile of attention deficits in young males with fragile X syndrome , 2000, Neuropsychologia.

[4]  Y. Benjamini,et al.  Controlling the false discovery rate: a practical and powerful approach to multiple testing , 1995 .

[5]  E. Berry-Kravis,et al.  Psychopharmacology in fragile X syndrome--present and future. , 2004, Mental retardation and developmental disabilities research reviews.

[6]  A. Kaiser,et al.  Parent and Teacher Agreement on Child Behavior Checklist Items in a Sample of Preschoolers From Low-Income and Predominantly African American Families , 2004, Journal of clinical child and adolescent psychology : the official journal for the Society of Clinical Child and Adolescent Psychology, American Psychological Association, Division 53.

[7]  A. Ouweland,et al.  Rapid antibody test for fragile X syndrome , 1995, The Lancet.

[8]  Deborah D. Hatton,et al.  Problem behavior in boys with fragile X syndrome. , 2002, American journal of medical genetics.

[9]  K. Gadow,et al.  A DSM-IV-referenced teacher rating scale for use in clinical management. , 2003, Journal of the American Academy of Child and Adolescent Psychiatry.

[10]  B. Oostra,et al.  FMRP expression as a potential prognostic indicator in fragile X syndrome. , 1999, American journal of medical genetics.

[11]  S Eliez,et al.  Brain anatomy, gender and IQ in children and adolescents with fragile X syndrome. , 2001, Brain : a journal of neurology.

[12]  A L Reiss,et al.  Specification of the neurobehavioral phenotype in males with fragile X syndrome. , 1995, Pediatrics.

[13]  Allan L Reiss,et al.  The neuroanatomy and neuroendocrinology of fragile X syndrome. , 2004, Mental retardation and developmental disabilities research reviews.

[14]  J. Fryns,et al.  Fragile (X) syndrome: a study of the psychological profile in 23 prepubertal patients , 1987, Clinical genetics.

[15]  Deborah D. Hatton,et al.  Adaptive behavior in children with fragile X syndrome. , 2003, American journal of mental retardation : AJMR.

[16]  A. Reiss,et al.  Molecular and cellular genetics of fragile X syndrome. , 1999, American journal of medical genetics.

[17]  R. Hagerman,et al.  A controlled trial of stimulant medication in children with the fragile X syndrome. , 1988, American journal of medical genetics.

[18]  W. Kaufmann,et al.  Social behavior profile in young males with fragile X syndrome: Characteristics and specificity , 2004, American journal of medical genetics. Part A.

[19]  F. Munir,et al.  Differential Impact of the FMR-1 Full Mutation on Memory and Attention Functioning: A Neuropsychological Perspective , 2001, Journal of Cognitive Neuroscience.

[20]  P. Jin,et al.  RNA and microRNAs in fragile X mental retardation , 2004, Nature Cell Biology.

[21]  K. Cornish,et al.  Attention and language in fragile X. , 2004, Mental retardation and developmental disabilities research reviews.

[22]  M. Rice,et al.  Stability of behavioral ratings of children with SLI. , 2002, Journal of speech, language, and hearing research : JSLHR.

[23]  D. Lane,et al.  Treatment effects of methylphenidate on behavioral adjustment in children with mental retardation and ADHD. , 2003, Journal of the American Academy of Child and Adolescent Psychiatry.

[24]  H. Berghe,et al.  The psychological profile of the fragile X syndrome , 1984, Clinical genetics.

[25]  M. Mazzocco,et al.  Advances in research on the fragile X syndrome. , 2000, Mental retardation and developmental disabilities research reviews.

[26]  R I Richards,et al.  Mapping of DNA instability at the fragile X to a trinucleotide repeat sequence p(CCG)n , 1991, Science.

[27]  R. Doherty Long term follow up , 1999, BMJ.

[28]  T. Achenbach Manual for the child behavior checklist/4-18 and 1991 profile , 1991 .

[29]  A. Gontard,et al.  Intelligence and cognitive function in children and adolescents with spinal muscular atrophy , 2002, Neuromuscular Disorders.

[30]  S. Lévesque,et al.  Premutation and intermediate-size FMR1 alleles in 10572 males from the general population: loss of an AGG interruption is a late event in the generation of fragile X syndrome alleles. , 2002, Human molecular genetics.

[31]  J. Bregman,et al.  Fragile X syndrome: genetic predisposition to psychopathology , 1988, Journal of autism and developmental disorders.

[32]  L. Freund,et al.  Early behavior signs of psychiatric phenotypes in fragile X syndrome. , 2000, American journal of mental retardation : AJMR.

[33]  B. Tonge,et al.  Longitudinal course of behavioral and emotional problems in fragile X syndrome. , 1999, American journal of medical genetics.

[34]  J. Turk Fragile X Syndrome and Attentional Deficits , 1998 .

[35]  J. Wilding,et al.  Further delineation of the executive deficit in males with fragile-X syndrome , 2002, Neuropsychologia.

[36]  I. Weiler,et al.  Fragile X mental retardation protein is translated near synapses in response to neurotransmitter activation. , 1997, Proceedings of the National Academy of Sciences of the United States of America.

[37]  Subcommittee on Attention-Deficit,et al.  Clinical Practice Guideline: Diagnosis and Evaluation of the Child With Attention-Deficit/Hyperactivity Disorder , 2000 .

[38]  D. Dawson,et al.  Behavior problems of young girls with fragile X syndrome: factor scores on the Conners' Parent's Questionnaire. , 1994, American journal of medical genetics.

[39]  Deborah D. Hatton,et al.  Variability in FMRP and early development in males with fragile X syndrome. , 2001, American journal of mental retardation : AJMR.

[40]  A. Reiss,et al.  Psychiatric disorders associated with fragile X in the young female. , 1993, Pediatrics.

[41]  P. Mirrett,et al.  Early Intervention Practices and Communication Intervention Strategies for Young Males With Fragile X Syndrome. , 2003, Language, speech, and hearing services in schools.

[42]  Anthony S. Bryk,et al.  Hierarchical Linear Models: Applications and Data Analysis Methods , 1992 .

[43]  A. Gontard,et al.  Cognitive and behavioral profile of fragile X boys: correlations to molecular data. , 2000, American journal of medical genetics.

[44]  A. Lachiewicz Abnormal behaviors of young girls with fragile X syndrome. , 1992, American journal of medical genetics.

[45]  J. Janosky,et al.  Long-Term Follow-Up of Children with Mental Retardation/Borderline Intellectual Functioning and ADHD , 1997, Journal of abnormal child psychology.

[46]  B. Tonge,et al.  Behavioural and emotional disturbance in fragile X syndrome. , 1994, American journal of medical genetics.

[47]  P. Rapiejko,et al.  Requirement of GTP hydrolysis for dissociation of the signal recognition particle from its receptor , 1991, Science.

[48]  Allan L. Reiss,et al.  Social Functioning Among Girls with Fragile X or Turner Syndrome and Their Sisters , 1998, Journal of autism and developmental disorders.

[49]  S. Faraone,et al.  Correspondence of parent and teacher reports in medication trials , 2005, European Child & Adolescent Psychiatry.

[50]  Jon Driver,et al.  Visual search in typically developing toddlers and toddlers with Fragile X or Williams syndrome. , 2004, Developmental science.

[51]  Deborah D. Hatton,et al.  Early developmental trajectories of males with fragile X syndrome. , 1998, American journal of mental retardation : AJMR.

[52]  Susan A. Sullivan,et al.  Leiter International Performance Scale–Revised , 1998 .

[53]  R. Hagerman,et al.  Medication use in fragile X syndrome , 2001 .

[54]  Dana C Crawford,et al.  FMR1 and the fragile X syndrome: Human genome epidemiology review , 2001, Genetics in Medicine.