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. 2024 May 27;32(9):1166–1183. doi: 10.1038/s41431-024-01616-9

Table 2.

Result of SF analysis and specific questionnaires at TR, T6, and T12.

Index Cases SF1 (n = 16/18 returned) SF2 (n = 20/22 returned) SF3 (n = 11/13 returned)
Name of gene involved

Cardiac: DSG2 (1)a, FBN1 (1)a, MYBPC3 (1)a, MYH7 (1)a, RYR2b, TNNI3 (1)a

Cancer: BRCA2 (1)a, SDHB (1)a

Metabolic: LDLR (3)a,c, PCSK9 (1)

Bleeding disorder: PROC (1)a, SERPINC1 (1)a, VWF (2)a

Other: COL3A1b (1) DMD (1)a

CFTR (2)a

CYP21A2 (2)

HFE (15)a,c

SMN1 (1)a

Pseudogene to gene CYP21A2 and SMN1b

CYP2C19 Hmz (6)a,c

CYP2C9 Hmz (7 + 2b)a

Identification of primary finding (PF) With PF 7/16 (44%) 8/20 (40%) 3/11 (27%)
Without PF 9/16 (56%) 12/20 (60%) 8/11 (73%)
SPECIFIC QUESTIONNAIRE AT TR (only in responding parents) N = 19 N = 18 N = 14
Satisfaction to have SF results (fairly to fully) 89% 94% 100%
Worry (fairly to fully) 37% 35% 14%
Regret requesting access to SF 0% 0% 0%
SPECIFIC QUESTIONNAIRE AT T6 AND T12
Change in lifestyle habits for the child (a little to a lot) T6 6/16 (38%) 2/16 (13%) 1/10 (10%)
T12 6/13 (46%) 4/17 (24%) 2/11 (18%)
Implementation of some actions of prevention for the child (food, sport…) T6 7/16 (44%) 0/16 (0%) 1/10 (10%)
T12 5/13 (38%) 1/17 (6%) 2/11 (18%)
Change in future projects T6 0/16 (0%) 2/17 (12%) 0/10 (0%)
T12 3/13 (23%) 4/17 (24%) 1/10 (10%)
Change in the vision of child’s future T6 3/16 (19%) 4/16 (25%) 0/10 (0%)
T12 2/13 (15%) 3/17 (18%) 2/11 (18%)
Changes in perception of the child’s body T6 2/16 (13%) 1/16 (6%) 0/10 (0%)
T12 2/13 (15%) 0/17 (0%) 0/11 (0%)
Change in the relationship with their child T6 5/16 (31%) 1/16 (6%) 1/10 (10%)
T12 3/12 (25%) 4/17 (24%) 0/11 (0%)
Change in perception of their child T6 1/16 (6%) 2/16 (13%) 1/10 (10%)
T12 0/12 (0%) 0/17 (0%) 0/11 (0%)
SPECIFIC QUESTIONNAIRE AT T12
One year later, how do you feel about the SF results?
Satisfaction (fairly to fully) 5/5 (100%) 4/4 (100%) 2/3 (67%)
Worry (fairly to fully) 0/5 (0%) 0/4 (0%) 2/3 (67%)

Hmz homozgote, SF secondary findings, TR time at results, T6 time at 6 months after results, T12 time at 12 months after results.

aIncluded individuals in the qualitative part.

bNot returned to patient (change of mind or not back for consultation).

c2SF.