Assessing the Psychosocial Impacts of Whole-Genome Sequencing Outcomes on Orofacial Cleft Caregivers in Nigeria: A Mixed-Methods Study

TY - JOUR

T1 - Assessing the Psychosocial Impacts of Whole-Genome Sequencing Outcomes on Orofacial Cleft Caregivers in Nigeria

T2 - A Mixed-Methods Study

AU - Oladayo, Abimbola M.

AU - Sule, Veronica

AU - Oshodi, Yewande

AU - Adekunle, Adegbayi A.

AU - Adeyemo, Wasiu L.

AU - Ogunlewe, Oluwagbemiga

AU - Ayelomi, Oluwanifemi

AU - Babatunde, Adejoke

AU - Aikomo, Boluwatife

AU - Ajadi, Abosede

AU - Dabdoub, Shareef M.

AU - Pendleton, Chandler

AU - Busch, Tamara

AU - Alade, Azeez

AU - Olujitan, Mojisola

AU - Aladenika, Emmanuel

AU - Awotoye, Waheed

AU - Gowans, Lord J.J.

AU - Eshete, Mekonen

AU - Campbell, Colleen

AU - Caplan, Daniel J.

AU - Adeagbo, Oluwafemi

AU - Mossey, Peter A.

AU - Adeyemo, Adebowale A.

AU - Murray, Jeffrey C.

AU - Prince, Anya E.R.

AU - Butali, Azeez

N1 - Publisher Copyright: © 2025, American Cleft Palate Craniofacial Association.

PY - 2025/4/13

Y1 - 2025/4/13

N2 - Objective: 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.

AB - Objective: 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.

KW - ELSI

KW - mental health

KW - Nigeria

KW - orofacial clefts

KW - psychosocial impact

KW - transactional theory of stress and coping

U2 - 10.1177/10556656251332351

DO - 10.1177/10556656251332351

M3 - Article

C2 - 40223298

AN - SCOPUS:105002962954

SN - 1055-6656

JO - Cleft Palate Craniofacial Journal

JF - Cleft Palate Craniofacial Journal

ER -