To investigate the behavioral outcomes of children with clefts and the psychosocial impact and mental health experiences of their caregivers regarding unintended outcomes of genomic sequencing, that is, secondary genetic findings (SFs).
Design
Convergent parallel mixed methods.
Setting
The cleft and immunization clinics at the Lagos University Teaching, Hospital, Nigeria.
Participants
In total, 127 cases and 158 control caregiver child dyads (quantitative) and 22 caregivers of children with clefts (qualitative).
Main Outcome Measures
Standardized questionnaires were used to assess caregiver-reported mental health, quality of life (QoL), and their children's behavioral difficulties. In 2 focus groups, participants shared their perspectives on genomic testing outcomes, including SFs, mental health, and expectations in the context of clefts.
Results
Compared to the control group, caregivers of children with clefts reported poorer mental health, lower QoL and more behavioral difficulties in their children. Also, they believed that introducing genomic testing outcomes, including SFs, may exacerbate existing burdens. Following qualitative data analyses, 5 themes emerged—genetics knowledge/awareness, stressors, cognitive appraisals, coping/support strategies, and negative impact/positive gains. Both qualitative and quantitative results showed that caring for children with clefts required significant financial resources placing substantial stress on caregivers.
Conclusions
These findings highlight the importance of addressing caregivers’ psychosocial needs and the need for proactive measures to prepare for the return of genomic sequencing outcomes to patients and research participants, particularly in resource-limited settings like Africa, where such support may be lacking.
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