ScreenPlus Parental Attitudes on the Use of Residual Dried Blood Spots
Ethical Legal Social Issues (ELSI) Public Health and Policy
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Introduction:
Once newborn screening (NBS) testing is completed, the residual dried blood spots (DBS) can be stored and accessed for diagnostic purposes, quality improvement (QI), and public health research. Most programs store de-identified DBS which can be accessed and re-identified in rare circumstances. There are widely publicized concerns, however, about the use, storage, and protections of residual DBS and the role of parental informed consent. These concerns and resulting lawsuits have led to the destruction of millions of stored DBS. ScreenPlus is a pilot NBS program screening consented newborns for an additional panel of rare genetic disorders in New York City. A key objective is to assess the ethical, legal, and social implications (ELSI) of NBS practices and policies for complex conditions through a survey series and qualitative interviews. The latest survey focuses on capturing the nuances of parental perspectives on the uses of DBS and trust in various entities.
Methods:
Between May 2021 and October 2024, parents were invited to the ScreenPlus survey series via REDCap (N=7,928) and 3% completed the 25-item DBS survey (N=241). Six unique scenarios on ways DBS can be used were presented and parents were asked questions on level of support (4-point Likert), concerns and parental permission (yes/no). Responses were collapsed into binary outcomes ‘support’ and ‘oppose’ for each scenario. Trust in researchers and the state public health department (DOH) was assessed by 3 items (4-point Likert).
Results:
The majority of respondents were mothers (93%) aged 25-44 (90%) and self-reported as non-White (57%). Parents strongly supported the use of de-identified DBS in the scenarios presented. Specifically, 98% supported their use to develop screening for a disease with a new treatment, 97% to study genetics and disease risk, 96% for environmental research, and 94% for NBS QI. When it came to re-identified DBS, most parents supported their use by law enforcement to identify a missing child (92%), but fewer approved of their use to identify a potential criminal suspect (70%). Concerns about DBS use mirrored this trend: 45% expressed concerns about identifying criminal suspects, compared to 21% for identifying a missing child. Concerns were notably lower for other uses: 15% for NBS QI, 11% for environmental research and studying genetics and disease risk, and 10% for developing screening linked to new treatments. Across all scenarios, the majority of parents felt permission is necessary to use DBS (64-74%) and strongly recommended providing clear information on the protections of their child’s health data (71%). Parents had varying degrees of trust in their health system; 74% trust medical researchers, 62% state DOH, and 57% trust the state DOH will protect their child’s privacy.
Conclusion:
These findings suggest parents support the use of DBS for a wide range of uses, but feel parental permission is essential, regardless of its application. While trust varied in surveyed entities, parents expressed concerns about privacy and data protections against potential misuse. Implementing comprehensive educational resources and fostering transparent discussions about how their child's DBS is utilized and accessed can better inform parents. A potential limitation is parental bias as this cohort consented to participate in ScreenPlus. Effective NBS policies must address concerns related to public confidence and privacy, while striving to establish robust data protection practices and ensure transparency. By safeguarding parents’ autonomy within the healthcare system, these approaches will help strengthen and create a more accountable NBS system to meet the needs across all communities.
Once newborn screening (NBS) testing is completed, the residual dried blood spots (DBS) can be stored and accessed for diagnostic purposes, quality improvement (QI), and public health research. Most programs store de-identified DBS which can be accessed and re-identified in rare circumstances. There are widely publicized concerns, however, about the use, storage, and protections of residual DBS and the role of parental informed consent. These concerns and resulting lawsuits have led to the destruction of millions of stored DBS. ScreenPlus is a pilot NBS program screening consented newborns for an additional panel of rare genetic disorders in New York City. A key objective is to assess the ethical, legal, and social implications (ELSI) of NBS practices and policies for complex conditions through a survey series and qualitative interviews. The latest survey focuses on capturing the nuances of parental perspectives on the uses of DBS and trust in various entities.
Methods:
Between May 2021 and October 2024, parents were invited to the ScreenPlus survey series via REDCap (N=7,928) and 3% completed the 25-item DBS survey (N=241). Six unique scenarios on ways DBS can be used were presented and parents were asked questions on level of support (4-point Likert), concerns and parental permission (yes/no). Responses were collapsed into binary outcomes ‘support’ and ‘oppose’ for each scenario. Trust in researchers and the state public health department (DOH) was assessed by 3 items (4-point Likert).
Results:
The majority of respondents were mothers (93%) aged 25-44 (90%) and self-reported as non-White (57%). Parents strongly supported the use of de-identified DBS in the scenarios presented. Specifically, 98% supported their use to develop screening for a disease with a new treatment, 97% to study genetics and disease risk, 96% for environmental research, and 94% for NBS QI. When it came to re-identified DBS, most parents supported their use by law enforcement to identify a missing child (92%), but fewer approved of their use to identify a potential criminal suspect (70%). Concerns about DBS use mirrored this trend: 45% expressed concerns about identifying criminal suspects, compared to 21% for identifying a missing child. Concerns were notably lower for other uses: 15% for NBS QI, 11% for environmental research and studying genetics and disease risk, and 10% for developing screening linked to new treatments. Across all scenarios, the majority of parents felt permission is necessary to use DBS (64-74%) and strongly recommended providing clear information on the protections of their child’s health data (71%). Parents had varying degrees of trust in their health system; 74% trust medical researchers, 62% state DOH, and 57% trust the state DOH will protect their child’s privacy.
Conclusion:
These findings suggest parents support the use of DBS for a wide range of uses, but feel parental permission is essential, regardless of its application. While trust varied in surveyed entities, parents expressed concerns about privacy and data protections against potential misuse. Implementing comprehensive educational resources and fostering transparent discussions about how their child's DBS is utilized and accessed can better inform parents. A potential limitation is parental bias as this cohort consented to participate in ScreenPlus. Effective NBS policies must address concerns related to public confidence and privacy, while striving to establish robust data protection practices and ensure transparency. By safeguarding parents’ autonomy within the healthcare system, these approaches will help strengthen and create a more accountable NBS system to meet the needs across all communities.