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Psychological well-being of mothers of youth with fragile X syndrome: syndrome specificity and within-syndrome variability.
J Intellect Disabil Res. 2006 Dec; 50(Pt 12):894-904.JI

Abstract

BACKGROUND

Research on parental well-being has focused largely on Down syndrome and autism; however, fragile X syndrome is likely to pose different challenges for parents compared with these other diagnostic conditions. Moreover, there is considerable variability among youth with fragile X syndrome; for example, 25% to 33% of affected youth meet criteria for a co-morbid diagnosis of autism. It is likely that parents of youth with fragile X syndrome will experience different degrees and patterns of stress, depending on whether their offspring do or do not have a co-morbid diagnosis of autism. In the present study, we compared mothers of three groups of young males on measures of psychological well-being and stress: those with fragile X syndrome and a co-morbid diagnosis of autism; those with fragile X syndrome alone; and those with Down syndrome.

METHOD

The sample consisted of mothers of adolescent and young adult males with fragile X syndrome and co-morbid autism (n=9), fragile X syndrome alone (n=19), and Down syndrome (n=19). We screened all youth for autism using the Autism Behavior Checklist, which was completed by mothers, fathers and teachers, and the youth who scored above the suggested cut-off were evaluated by a licensed psychologist to determine autism status. The three groups of youth did not differ in chronological age (16.4, 15.8 and 16.0 years, respectively) or non-verbal mental age (3.8, 3.9 and 3.8 years, respectively). Several self-report measures were completed by mothers. These measures assessed current mental health status (e.g. the Center for Epidemiological Studies Depression Scale), perceptions of their son's and family's functioning (e.g. the Positive Affect Index, which measures closeness felt by the mother to her son and also reciprocated closeness felt by the son towards the mother, as perceived by the mother), and approach to coping with their son's disability [e.g. the Multidimensional Coping Inventory (COPE), which measures emotion-focused and problem-solving focused coping].

RESULTS

The results suggest that fragile X syndrome creates more challenges to maternal psychological well-being than Down syndrome, and that the combination of fragile X syndrome and autism can be particularly challenging. Differences among groups, however, were manifested mainly as concerns about the affected son and about relationships within the family rather than as lower levels of mental health. Thus, mothers of sons with fragile X syndrome, regardless of the son's autism status, reported more pessimism about the son's future and more conflict within the family than mothers of sons with Down syndrome. Additionally, mothers of sons with fragile X syndrome and co-morbid autism reported lower levels of reciprocated closeness than the other two groups of mothers.

CONCLUSION

We consider possible causes of these maternal differences, the implications for clinical practice, needs for future research, and the importance of understanding child and contextual factors as well as the dynamics leading to these differences.

Authors+Show Affiliations

Waisman Center, University of Wisconsin-Madison, WI, USA. lewis@waisman.wisc.eduNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article
Research Support, N.I.H., Extramural

Language

eng

PubMed ID

17100950

Citation

Lewis, P, et al. "Psychological Well-being of Mothers of Youth With Fragile X Syndrome: Syndrome Specificity and Within-syndrome Variability." Journal of Intellectual Disability Research : JIDR, vol. 50, no. Pt 12, 2006, pp. 894-904.
Lewis P, Abbeduto L, Murphy M, et al. Psychological well-being of mothers of youth with fragile X syndrome: syndrome specificity and within-syndrome variability. J Intellect Disabil Res. 2006;50(Pt 12):894-904.
Lewis, P., Abbeduto, L., Murphy, M., Richmond, E., Giles, N., Bruno, L., Schroeder, S., Anderson, J., & Orsmond, G. (2006). Psychological well-being of mothers of youth with fragile X syndrome: syndrome specificity and within-syndrome variability. Journal of Intellectual Disability Research : JIDR, 50(Pt 12), 894-904.
Lewis P, et al. Psychological Well-being of Mothers of Youth With Fragile X Syndrome: Syndrome Specificity and Within-syndrome Variability. J Intellect Disabil Res. 2006;50(Pt 12):894-904. PubMed PMID: 17100950.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Psychological well-being of mothers of youth with fragile X syndrome: syndrome specificity and within-syndrome variability. AU - Lewis,P, AU - Abbeduto,L, AU - Murphy,M, AU - Richmond,E, AU - Giles,N, AU - Bruno,L, AU - Schroeder,S, AU - Anderson,J, AU - Orsmond,G, PY - 2006/11/15/pubmed PY - 2007/2/21/medline PY - 2006/11/15/entrez SP - 894 EP - 904 JF - Journal of intellectual disability research : JIDR JO - J Intellect Disabil Res VL - 50 IS - Pt 12 N2 - BACKGROUND: Research on parental well-being has focused largely on Down syndrome and autism; however, fragile X syndrome is likely to pose different challenges for parents compared with these other diagnostic conditions. Moreover, there is considerable variability among youth with fragile X syndrome; for example, 25% to 33% of affected youth meet criteria for a co-morbid diagnosis of autism. It is likely that parents of youth with fragile X syndrome will experience different degrees and patterns of stress, depending on whether their offspring do or do not have a co-morbid diagnosis of autism. In the present study, we compared mothers of three groups of young males on measures of psychological well-being and stress: those with fragile X syndrome and a co-morbid diagnosis of autism; those with fragile X syndrome alone; and those with Down syndrome. METHOD: The sample consisted of mothers of adolescent and young adult males with fragile X syndrome and co-morbid autism (n=9), fragile X syndrome alone (n=19), and Down syndrome (n=19). We screened all youth for autism using the Autism Behavior Checklist, which was completed by mothers, fathers and teachers, and the youth who scored above the suggested cut-off were evaluated by a licensed psychologist to determine autism status. The three groups of youth did not differ in chronological age (16.4, 15.8 and 16.0 years, respectively) or non-verbal mental age (3.8, 3.9 and 3.8 years, respectively). Several self-report measures were completed by mothers. These measures assessed current mental health status (e.g. the Center for Epidemiological Studies Depression Scale), perceptions of their son's and family's functioning (e.g. the Positive Affect Index, which measures closeness felt by the mother to her son and also reciprocated closeness felt by the son towards the mother, as perceived by the mother), and approach to coping with their son's disability [e.g. the Multidimensional Coping Inventory (COPE), which measures emotion-focused and problem-solving focused coping]. RESULTS: The results suggest that fragile X syndrome creates more challenges to maternal psychological well-being than Down syndrome, and that the combination of fragile X syndrome and autism can be particularly challenging. Differences among groups, however, were manifested mainly as concerns about the affected son and about relationships within the family rather than as lower levels of mental health. Thus, mothers of sons with fragile X syndrome, regardless of the son's autism status, reported more pessimism about the son's future and more conflict within the family than mothers of sons with Down syndrome. Additionally, mothers of sons with fragile X syndrome and co-morbid autism reported lower levels of reciprocated closeness than the other two groups of mothers. CONCLUSION: We consider possible causes of these maternal differences, the implications for clinical practice, needs for future research, and the importance of understanding child and contextual factors as well as the dynamics leading to these differences. SN - 0964-2633 UR - https://www.unboundmedicine.com/medline/citation/17100950/Psychological_well_being_of_mothers_of_youth_with_fragile_X_syndrome:_syndrome_specificity_and_within_syndrome_variability_ L2 - https://doi.org/10.1111/j.1365-2788.2006.00907.x DB - PRIME DP - Unbound Medicine ER -