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Impact of genetic counseling and Connexin-26 and Connexin-30 testing on deaf identity and comprehension of genetic test results in a sample of deaf adults: a prospective, longitudinal study.

Palmer CG, Boudreault P, Baldwin EE, Sinsheimer JS - PLoS ONE (2014)

Bottom Line: This study specifically evaluated the effect of genetic counseling and Connexin-26 and Connexin-30 genetic test results on participants' deaf identity and understanding of their genetic test results.Four deaf identity orientations (hearing, marginal, immersion, bicultural) were evaluated using subscales of the Deaf Identity Development Scale-Revised.We found evidence that participants understood their specific genetic test results following genetic counseling, but found no evidence of change in deaf identity based on genetic counseling or their genetic test results.

View Article: PubMed Central - PubMed

Affiliation: Department of Psychiatry & Biobehavioral Sciences, University of California Los Angeles, Los Angeles, California, United States of America; Department of Human Genetics, University of California Los Angeles, Los Angeles, California, United States of America; Institute for Society and Genetics, University of California Los Angeles, Los Angeles, California, United States of America.

ABSTRACT
Using a prospective, longitudinal study design, this paper addresses the impact of genetic counseling and testing for deafness on deaf adults and the Deaf community. This study specifically evaluated the effect of genetic counseling and Connexin-26 and Connexin-30 genetic test results on participants' deaf identity and understanding of their genetic test results. Connexin-26 and Connexin-30 genetic testing was offered to participants in the context of linguistically and culturally appropriate genetic counseling. Questionnaire data collected from 209 deaf adults at four time points (baseline, immediately following pre-test genetic counseling, 1-month following genetic test result disclosure, and 6-months after result disclosure) were analyzed. Four deaf identity orientations (hearing, marginal, immersion, bicultural) were evaluated using subscales of the Deaf Identity Development Scale-Revised. We found evidence that participants understood their specific genetic test results following genetic counseling, but found no evidence of change in deaf identity based on genetic counseling or their genetic test results. This study demonstrated that culturally and linguistically appropriate genetic counseling can improve deaf clients' understanding of genetic test results, and the formation of deaf identity was not directly related to genetic counseling or Connexin-26 and Connexin-30 genetic test results.

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2A–2D: DIDS-R subscale agreement score least squares estimates by Connexin result group before and after genetic testing.A. hearing subscale, main effects: Connexin p = 0.84, time p = 0.08; interaction: Connexin x time p = 0.02. Non-significant pairwise comparisons at α = 0.05 at each timepoint. B. marginal subscale, main effects: Connexin p = 0.45, time p = 0.61; interaction: Connexin x time p = 0.25. C. immersion subscale, main effects: Connexin p = 0.32, time p = 0.32; interaction: Connexin x time p = 0.20. D. bicultural subscale, main effects: Connexin p = 0.88, time p = 0.23; interaction: Connexin x time p = 0.27. 1 = strongly agree, 5 = strongly disagree. Cx = connexin; GC = genetic counseling; M = month.
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pone-0111512-g002: 2A–2D: DIDS-R subscale agreement score least squares estimates by Connexin result group before and after genetic testing.A. hearing subscale, main effects: Connexin p = 0.84, time p = 0.08; interaction: Connexin x time p = 0.02. Non-significant pairwise comparisons at α = 0.05 at each timepoint. B. marginal subscale, main effects: Connexin p = 0.45, time p = 0.61; interaction: Connexin x time p = 0.25. C. immersion subscale, main effects: Connexin p = 0.32, time p = 0.32; interaction: Connexin x time p = 0.20. D. bicultural subscale, main effects: Connexin p = 0.88, time p = 0.23; interaction: Connexin x time p = 0.27. 1 = strongly agree, 5 = strongly disagree. Cx = connexin; GC = genetic counseling; M = month.

Mentions: Repeated measures regression analysis with covariates was then performed to determine if participants' responses to these deaf identity subscales changed over the course of the study, particularly after they learned their genetic test results. Figures 2A–2D plot participants' least square estimates of the DIDS-R subscale scores by Connexin result group before and after receiving their genetic test results; and Table S1 provides the least square estimates and standard errors for the main effects, covariates, and interaction term. Regardless of Connexin result, endorsement of the bicultural subscale was strongest, and endorsement of the hearing and marginal subscales was weakest, at each time point.


Impact of genetic counseling and Connexin-26 and Connexin-30 testing on deaf identity and comprehension of genetic test results in a sample of deaf adults: a prospective, longitudinal study.

Palmer CG, Boudreault P, Baldwin EE, Sinsheimer JS - PLoS ONE (2014)

2A–2D: DIDS-R subscale agreement score least squares estimates by Connexin result group before and after genetic testing.A. hearing subscale, main effects: Connexin p = 0.84, time p = 0.08; interaction: Connexin x time p = 0.02. Non-significant pairwise comparisons at α = 0.05 at each timepoint. B. marginal subscale, main effects: Connexin p = 0.45, time p = 0.61; interaction: Connexin x time p = 0.25. C. immersion subscale, main effects: Connexin p = 0.32, time p = 0.32; interaction: Connexin x time p = 0.20. D. bicultural subscale, main effects: Connexin p = 0.88, time p = 0.23; interaction: Connexin x time p = 0.27. 1 = strongly agree, 5 = strongly disagree. Cx = connexin; GC = genetic counseling; M = month.
© Copyright Policy
Related In: Results  -  Collection

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Show All Figures
getmorefigures.php?uid=PMC4222828&req=5

pone-0111512-g002: 2A–2D: DIDS-R subscale agreement score least squares estimates by Connexin result group before and after genetic testing.A. hearing subscale, main effects: Connexin p = 0.84, time p = 0.08; interaction: Connexin x time p = 0.02. Non-significant pairwise comparisons at α = 0.05 at each timepoint. B. marginal subscale, main effects: Connexin p = 0.45, time p = 0.61; interaction: Connexin x time p = 0.25. C. immersion subscale, main effects: Connexin p = 0.32, time p = 0.32; interaction: Connexin x time p = 0.20. D. bicultural subscale, main effects: Connexin p = 0.88, time p = 0.23; interaction: Connexin x time p = 0.27. 1 = strongly agree, 5 = strongly disagree. Cx = connexin; GC = genetic counseling; M = month.
Mentions: Repeated measures regression analysis with covariates was then performed to determine if participants' responses to these deaf identity subscales changed over the course of the study, particularly after they learned their genetic test results. Figures 2A–2D plot participants' least square estimates of the DIDS-R subscale scores by Connexin result group before and after receiving their genetic test results; and Table S1 provides the least square estimates and standard errors for the main effects, covariates, and interaction term. Regardless of Connexin result, endorsement of the bicultural subscale was strongest, and endorsement of the hearing and marginal subscales was weakest, at each time point.

Bottom Line: This study specifically evaluated the effect of genetic counseling and Connexin-26 and Connexin-30 genetic test results on participants' deaf identity and understanding of their genetic test results.Four deaf identity orientations (hearing, marginal, immersion, bicultural) were evaluated using subscales of the Deaf Identity Development Scale-Revised.We found evidence that participants understood their specific genetic test results following genetic counseling, but found no evidence of change in deaf identity based on genetic counseling or their genetic test results.

View Article: PubMed Central - PubMed

Affiliation: Department of Psychiatry & Biobehavioral Sciences, University of California Los Angeles, Los Angeles, California, United States of America; Department of Human Genetics, University of California Los Angeles, Los Angeles, California, United States of America; Institute for Society and Genetics, University of California Los Angeles, Los Angeles, California, United States of America.

ABSTRACT
Using a prospective, longitudinal study design, this paper addresses the impact of genetic counseling and testing for deafness on deaf adults and the Deaf community. This study specifically evaluated the effect of genetic counseling and Connexin-26 and Connexin-30 genetic test results on participants' deaf identity and understanding of their genetic test results. Connexin-26 and Connexin-30 genetic testing was offered to participants in the context of linguistically and culturally appropriate genetic counseling. Questionnaire data collected from 209 deaf adults at four time points (baseline, immediately following pre-test genetic counseling, 1-month following genetic test result disclosure, and 6-months after result disclosure) were analyzed. Four deaf identity orientations (hearing, marginal, immersion, bicultural) were evaluated using subscales of the Deaf Identity Development Scale-Revised. We found evidence that participants understood their specific genetic test results following genetic counseling, but found no evidence of change in deaf identity based on genetic counseling or their genetic test results. This study demonstrated that culturally and linguistically appropriate genetic counseling can improve deaf clients' understanding of genetic test results, and the formation of deaf identity was not directly related to genetic counseling or Connexin-26 and Connexin-30 genetic test results.

Show MeSH
Related in: MedlinePlus