Dorn M B, Mazzocco M M, Hagerman R J
Child Development Unit, Children's Hospital, Denver, CO 80218.
J Am Acad Child Adolesc Psychiatry. 1994 Feb;33(2):256-64. doi: 10.1097/00004583-199402000-00015.
In the present study we examined the incidence of psychiatric and behavioral problems among male carriers of the fragile X gene.
Retrospective data on carrier males were gathered using the family informant method. Each of 56 fragile X carrier women was interviewed about her father by an examiner blind to the father's carrier status. The interviewer administered measures of (1) behaviors related to DSM-III-R Axis I disorders, (2) adult attention-deficit hyperactivity disorder (ADHD) behaviors, (3) parental bonding skills, and (4) abusive behaviors. The endorsements from 24 women with fragile X fathers were compared with endorsements from 32 women with nonfragile X fathers.
The results show a higher incidence of psychopathology among the fragile X males (relative to nonfragile X fathers) for behaviors related to adult ADHD, parental bonding, abuse; and particularly for alcohol abuse/dependence and obsessive-compulsive disorder behaviors.
These findings support the hypotheses that some "nonpenetrant" males may indeed be mildly affected carriers and that there is a broad spectrum of involvement among carrier males.
