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1.
Am J Med Genet ; 33(4): 513-8, 1989 Aug.
Article in English | MEDLINE | ID: mdl-2596514

ABSTRACT

Previous studies have reported a drop of IQ in males with Martin-Bell or fragile X syndrome during childhood and adolescence. It is uncertain when and why this drop occurs and whether it affects all fragile X males. We have analyzed longitudinal IQ data on all 24 fragile X males who have been followed for at least 2 years through the Child Development Unit in Denver and who have been evaluated with a Stanford-Binet Form L-M assessment. A significant drop in IQ occurred for the group as a whole, but an individual z analysis demonstrated that only 7 of 24 had a significant decline in IQ. Visual inspection demonstrates an initial decline in IQ beginning in middle childhood and continuing through adolescence. We postulate that this drop occurs because of relatively greater weaknesses with abstract reasoning and higher symbolic language skills that are stressed in the cognitive testing of later childhood and adolescence.


Subject(s)
Fragile X Syndrome/psychology , Intelligence , Sex Chromosome Aberrations/psychology , Adult , Humans , Longitudinal Studies , Male , Stanford-Binet Test
2.
Am J Med Genet ; 30(1-2): 377-92, 1988.
Article in English | MEDLINE | ID: mdl-3052064

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.


Subject(s)
Dextroamphetamine/therapeutic use , Fragile X Syndrome/drug therapy , Methylphenidate/therapeutic use , Sex Chromosome Aberrations/drug therapy , Attention Deficit Disorder with Hyperactivity/drug therapy , Attention Deficit Disorder with Hyperactivity/etiology , Child , Child, Preschool , Clinical Trials as Topic , Double-Blind Method , Female , Fragile X Syndrome/complications , Fragile X Syndrome/psychology , Humans , Male
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