How does the mode of inheritance of a genetic condition influence families? A study of guilt, blame, stigma, and understanding of inheritance and reproductive risks in families with X-linked and autosomal recessive diseases

Cynthia Anne James, Donald W. Hadley, Neil A. Holtzman, Jerry A. Winkelstein

Research output: Contribution to journalArticle

Abstract

Purpose: While the mode of inheritance of a genetic condition has long been considered to have not only medical, but also psychosocial consequences for families, this supposition has never been tested. Methods: We surveyed 112 members of 51 families (59% response) with chronic granulomatous disease to determine the influence of mode of inheritance on parents', siblings', and patients' (1) knowledge of inheritance and reproductive risk; (2) concern about risk to future family-members; (3) feelings of guilt and blame; and (4) feelings of stigmatization. Ninety-six members of 51 families (49% response) with Duchenne/Becker muscular dystrophy and spinal muscular atrophy types II/III were also studied. Results: X-linked families had better understanding of inheritance (P <0.001) and reproductive risks (P <0.01). X-linked mothers worried more about risks to future generations; other autosomal-recessive family members were as worried. X-linked mothers were more likely to feel guilty (P <0.01) and blame themselves (P <0.001). X-linked fathers blamed their child's mother (P <0.05) and X-linked mothers felt more blamed by the father (P <0.01). X-linked family-members were more likely to consider being a carrier stigmatizing (P <0.05). Conclusion: When providing genetic counseling, attention should be given to guilt and blame in X-linked families and understanding reproductive risks in autosomal recessive families.

Original languageEnglish (US)
Pages (from-to)234-242
Number of pages9
JournalGenetics in Medicine
Volume8
Issue number4
DOIs
StatePublished - Apr 2006

Fingerprint

Guilt
Mothers
Fathers
Emotions
Spinal Muscular Atrophies of Childhood
Chronic Granulomatous Disease
Stereotyping
Duchenne Muscular Dystrophy
Social Responsibility
Genetic Counseling
Siblings
Parents

Keywords

  • Chronic granulomatous disease
  • Genetic counseling
  • Guilt
  • Knowledge
  • Stigma

ASJC Scopus subject areas

  • Genetics(clinical)
  • Genetics

Cite this

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title = "How does the mode of inheritance of a genetic condition influence families? A study of guilt, blame, stigma, and understanding of inheritance and reproductive risks in families with X-linked and autosomal recessive diseases",
abstract = "Purpose: While the mode of inheritance of a genetic condition has long been considered to have not only medical, but also psychosocial consequences for families, this supposition has never been tested. Methods: We surveyed 112 members of 51 families (59{\%} response) with chronic granulomatous disease to determine the influence of mode of inheritance on parents', siblings', and patients' (1) knowledge of inheritance and reproductive risk; (2) concern about risk to future family-members; (3) feelings of guilt and blame; and (4) feelings of stigmatization. Ninety-six members of 51 families (49{\%} response) with Duchenne/Becker muscular dystrophy and spinal muscular atrophy types II/III were also studied. Results: X-linked families had better understanding of inheritance (P <0.001) and reproductive risks (P <0.01). X-linked mothers worried more about risks to future generations; other autosomal-recessive family members were as worried. X-linked mothers were more likely to feel guilty (P <0.01) and blame themselves (P <0.001). X-linked fathers blamed their child's mother (P <0.05) and X-linked mothers felt more blamed by the father (P <0.01). X-linked family-members were more likely to consider being a carrier stigmatizing (P <0.05). Conclusion: When providing genetic counseling, attention should be given to guilt and blame in X-linked families and understanding reproductive risks in autosomal recessive families.",
keywords = "Chronic granulomatous disease, Genetic counseling, Guilt, Knowledge, Stigma",
author = "James, {Cynthia Anne} and Hadley, {Donald W.} and Holtzman, {Neil A.} and Winkelstein, {Jerry A.}",
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AU - James, Cynthia Anne

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AU - Holtzman, Neil A.

AU - Winkelstein, Jerry A.

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N2 - Purpose: While the mode of inheritance of a genetic condition has long been considered to have not only medical, but also psychosocial consequences for families, this supposition has never been tested. Methods: We surveyed 112 members of 51 families (59% response) with chronic granulomatous disease to determine the influence of mode of inheritance on parents', siblings', and patients' (1) knowledge of inheritance and reproductive risk; (2) concern about risk to future family-members; (3) feelings of guilt and blame; and (4) feelings of stigmatization. Ninety-six members of 51 families (49% response) with Duchenne/Becker muscular dystrophy and spinal muscular atrophy types II/III were also studied. Results: X-linked families had better understanding of inheritance (P <0.001) and reproductive risks (P <0.01). X-linked mothers worried more about risks to future generations; other autosomal-recessive family members were as worried. X-linked mothers were more likely to feel guilty (P <0.01) and blame themselves (P <0.001). X-linked fathers blamed their child's mother (P <0.05) and X-linked mothers felt more blamed by the father (P <0.01). X-linked family-members were more likely to consider being a carrier stigmatizing (P <0.05). Conclusion: When providing genetic counseling, attention should be given to guilt and blame in X-linked families and understanding reproductive risks in autosomal recessive families.

AB - Purpose: While the mode of inheritance of a genetic condition has long been considered to have not only medical, but also psychosocial consequences for families, this supposition has never been tested. Methods: We surveyed 112 members of 51 families (59% response) with chronic granulomatous disease to determine the influence of mode of inheritance on parents', siblings', and patients' (1) knowledge of inheritance and reproductive risk; (2) concern about risk to future family-members; (3) feelings of guilt and blame; and (4) feelings of stigmatization. Ninety-six members of 51 families (49% response) with Duchenne/Becker muscular dystrophy and spinal muscular atrophy types II/III were also studied. Results: X-linked families had better understanding of inheritance (P <0.001) and reproductive risks (P <0.01). X-linked mothers worried more about risks to future generations; other autosomal-recessive family members were as worried. X-linked mothers were more likely to feel guilty (P <0.01) and blame themselves (P <0.001). X-linked fathers blamed their child's mother (P <0.05) and X-linked mothers felt more blamed by the father (P <0.01). X-linked family-members were more likely to consider being a carrier stigmatizing (P <0.05). Conclusion: When providing genetic counseling, attention should be given to guilt and blame in X-linked families and understanding reproductive risks in autosomal recessive families.

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