Supplemental Table I. Non-significant relationships with parental concern
Factor / Strongly Disagree/ Disagreen (%) / Neutral
n (%) / Agree/Strongly Agree
n (%) / p-value
Parental Concern – Worried about Tumor Development
Location Groups
United States
Other / 25/173 (14.5%)
6/83 (7.2%) / 23/173 (13.3%)
14/83 (16.9%) / 125/173 (72.3%)
63/83 (75.9%) / 0.222
Genetic Cause
IC2 LOM
Other / 9/77 (11.7%)
13/98 (13.3%) / 14/77 (18.2%)
19/98 (19.4%) / 54/77 (70.1%)
66/98 (67.3%) / 0.920
Education Groups
Associate’s degree or less
Bachelor’s degree
Advanced degree / 12/96 (12.5%)
13/83 (15.7%)
6/69 (8.7%) / 11/96 (11.5%)
11/83 (13.3%)
12/69 (17.4%) / 73/96 (76.0%)
59/83 (71.1%)
51/69 (73.9%) / 0.616
Employment Groups
Unemployed or Homemaker
Part-Time
Full-Time / 8/78 (10.3%)
6/41 (14.6%)
17/125 (13.6%) / 8/78 (10.3%)
7/41 (17.1%)
21/125 (16.8%) / 62/78 (79.5%)
28/41 (68.3%)
87/125 (69.6%) / 0.573
Martial Status
Living alone
Living with someone / 4/32 (12.5%)
27/224 (12.1%) / 3/32 (9.4%)
35/224 (15.6%) / 25/32 (78.1%)
162/224 (72.3%) / 0.647
Incurred Costs
No
Yes / 17/154 (11.0%)
14/104 (13.5%) / 27/154 (17.5%)
13/104 (12.5%) / 110/154 (71.4%)
77/104 (74.0%) / 0.503
History of false-positive screen
No
Yes / 19/176 (10.8%)
12/83 (14.5%) / 26/176 (14.8%)
14/83 (16.9%) / 131/176 (74.4%)
57/83 (68.7%) / 0.592
Risk threshold to screen
Any risk
1-5%
6-10% / 19/187 (10.2%)
7/46 (15.2%)
3/14 (21.4%) / 24/187 (12.8%)
10/46 (21.7%)
3/14 (21.4%) / 144/187 (77.0%)
29/46 (63.0%)
8/14 (57.1%) / 0.209
Vary screening by type
No
Yes / 24/195 (12.3%)
7/59 (11.9%) / 28/195 (14.4%)
11/59 (18.6%) / 143/195 (73.3%)
41/59 (69.5%) / 0.726
Satisfaction with physician recommendations
No
Yes / 2/29 (6.9%)
29/229 (12.7%) / 3/29 (10.3%)
37/229 (16.2%) / 24/29 (82.8%)
163/229 (71.2%) / 0.417
Advocate to receive screening regimen
No
Yes / 23/153 (15.0%)
8/105 (7.6%) / 21/153 (13.7%)
19/105 (18.1%) / 109/153 (71.2%)
78/105 (74.3%) / 0.158
AFP frequency followed
6 weeks
3 months / 8/107 (7.5%)
17/101 (16.8%) / 18/107 (16.8%)
13/101 (12.9%) / 81/107 (75.7%)
71/101 (70.3%) / 0.104
AFP frequency recommended
6 weeks
3 months / 7/98 (7.1%)
20/119 (16.8%) / 15/98 (15.3%)
16/119 (13.4%) / 76/98 (77.6%)
83/119 (69.7%) / 0.100
AFP frequency beliefs
6 weeks
3 months / 12/140 (8.6%)
10/78 (12.8%) / 22/140 (15.7%)
12/78 (15.4%) / 106/140 (75.7%)
56/78 (71.8%) / 0.606
AFP frequency adherence
No
Yes / 25/207 (12.1%)
3/43 (7.0%) / 30/207 (14.5%)
10/43 (23.3%) / 152/207 (73.4%)
30/43 (69.8%) / 0.272
Knowledge
Incorrect
Correct / 5/66 (7.6%)
7/56 (12.5%) / 9/66 (13.6%)
13/56 (23.2%) / 52/66 (78.8%)
36/56 (64.3%) / 0.205
Parental Concern – Screening Helps Decrease Worry
Location Groups
United States
Other / 12/172 (7.0%)
6/83 (7.0%) / 18/172 (10.5%)
9/83 (10.8%) / 142/172 (82.6%)
68/83 (81.9%) / 0.992
Genetic Cause
IC2 LOM
Other / 5/76 (6.6%)
5/98 (5.1%) / 9/76 (11.8%)
10/98 (10.2%) / 62/76 (81.6%)
83/98 (84.7%) / 0.853
Number of Children
1 child
2 or more children / 3/48 (6.3%)
15/206 (7.3%) / 7/48 (14.6%)
19/206 (9.2%) / 38/48 (79.2%)
172/206 (83.5%) / 0.538
Child age groups
Under 4 years
4 - 7.99 years
8 years and over / 15/163 (9.2%)
2/58 (3.4%)
1/37 (2.7%) / 15/163 (9.2%)
8/58 (13.8%)
4/37 (10.8%) / 133/163 (81.6%)
48/58 (82.8%)
32/37 (86.5%) / 0.391
Education Groups
Associate’s degree or less
Bachelor’s degree
Advanced degree / 6/96 (6.3%)
7/82 (8.5%)
5/69 (7.2%) / 12/96 (12.5%)
8/82 (9.8%)
4/69 (5.8%) / 78/96 (81.3%)
67/82 (81.7%)
60/69 (87.0%) / 0.671
Employment Groups
Unemployed or Homemaker
Part-Time
Full-Time / 5/78 (6.4%)
1/40 (2.5%)
11/125 (8.8%) / 6/78 (7.7%)
1/40 (2.5%)
19/125 (15.2%) / 67/78 (85.9%)
38/40 (95.0%)
95/125 (76.0%) / 0.066
Martial Status
Living alone
Living with someone / 2/32 (6.3%)
16/223 (7.2%) / 4/32 (12.5%)
23/223 (10.3%) / 26/32 (81.3%)
184/223 (82.5%) / 0.921
Incurred Costs
No
Yes / 12/153 (7.8%)
6/104 (5.8%) / 14/153 (9.2%)
13/104 (12.5%) / 127/153 (83.0%)
85/104 (81.7%) / 0.590
History of false-positive screen
No
Yes / 14/175 (8.0%)
4/83 (4.8%) / 16/175 (9.1%)
11/83 (13.3%) / 145/175 (82.9%)
68/83 (81.9%) / 0.421
Vary screening by type
No
Yes / 14/194 (7.2%)
4/59 (6.8%) / 16/194 (8.2%)
9/59 (15.3%) / 164/194 (84.5%)
46/59 (78.0%) / 0.287
Satisfaction with physician recommendations
No
Yes / 1/29 (3.4%)
17/228 (7.5%) / 1/29 (3.4%)
26/228 (11.4%) / 27/29 (93.1%)
185/228 (81.1%) / 0.274
Advocate to receive screening regimen
No
Yes / 11/152 (7.2%)
6/105 (5.7%) / 16/152 (10.5%)
11/105 (10.5%) / 125/152 (82.2%)
88/105 (83.8%) / 0.889
AFP frequency followed
6 weeks
3 months / 8/106 (7.5%)
7/101 (6.9%) / 9/106 (8.5%)
12/101 (11.9%) / 89/106 (84.0%)
82/101 (81.2%) / 0.718
AFP frequency recommended
6 weeks
3 months / 6/97 (6.2%)
10/119 (8.4%) / 10/97 (10.3%)
13/119 (10.9%) / 81/97 (83.5%)
96/119 (80.7%) / 0.808
AFP frequency beliefs
6 weeks
3 months / 10/139 (7.2%)
4/78 (5.1%) / 11/139 (7.9%)
9/78 (11.5%) / 118/139 (84.9%)
65/78 (83.3%) / 0.589
AFP frequency adherence
No
Yes / 16/206 (7.8%)
2/43 (4.7%) / 21/206 (10.2%)
4/43 (9.3%) / 169/206 (82.0%)
37/43 (86.0%) / 0.750
Knowledge
Incorrect
Correct / 4/65 (6.2%)
4/56 (7.1%) / 5/65 (7.7%)
7/56 (12.5%) / 56/65 (86.2%)
45/56 (80.4%) / 0.648
Parental Concern – Screening Increases Worry
Location Groups
United States
Other / 105/173 (60.7%)
56/83 (67.5%) / 35/173 (20.2%)
10/83 (12.0%) / 33/173 (19.1%)
17/83 (20.5%) / 0.272
Genetic Cause
IC2 LOM
Other / 50/77 (64.9%)
62/98 (63.3%) / 13/77 (16.9%)
19/98 (19.4%) / 14/77 (18.2%)
17/98 (17.3%) / 0.912
Number of Children
1 child
2 or more children / 29/49 (59.2%)
131/206 (63.6%) / 10/49 (20.4%)
35/206 (17.0%) / 10/49 (20.4%)
40/206 (19.4%) / 0.817
Child age groups
Under 4 years
4 - 7.99 years
8 years and over / 101/164 (61.6%)
34/58 (58.6%)
29/37 (78.4%) / 28/164 (17.1%)
12/58 (20.7%)
5/37 (13.5%) / 35/164 (21.3%)
12/58 (20.7%)
3/37 (8.1%) / 0.276
Education Groups
Associate’s degree or less
Bachelor’s degree
Advanced degree / 60/96 (62.5%)
56/83 (67.5%)
42/69 (60.9%) / 17/96 (17.7%)
12/83 (14.5%)
14/69 (20.3%) / 19/96 (19.8%)
15/83 (18.1%)
13/69 (18.8%) / 0.892
Employment Groups
Unemployed or Homemaker
Part-Time
Full-Time / 45/78 (57.7%)
31/41 (75.6%)
78/125 (62.4%) / 16/78 (20.5%)
5/41 (12.2%)
22/125 (17.6%) / 17/78 (21.8%)
5/41 (12.2%)
25/125 (20.0%) / 0.434
Martial Status
Living alone
Living with someone / 22/32 (68.8%)
139/224 (62.1%) / 3/32 (9.4%)
42/224 (18.8%) / 7/32 (21.9%)
43/224 (19.2%) / 0.427
Incurred Costs
No
Yes / 101/154 (65.6%)
62/104 (59.6%) / 23/154 (14.9%)
22/104 (21.2%) / 30/154 (19.5%)
20/104 (19.2%) / 0.421
History of false-positive screen
No
Yes / 115/176 (65.3%)
49/83 (59.0%) / 31/176 (17.6%)
14/83 (16.9%) / 30/176 (17.0%)
20/83 (24.1%) / 0.402
Risk threshold to screen
Any risk
1-5%
6-10% / 119/187 (63.6%)
30/46 (65.2%)
7/14 (50.0%) / 30/187 (16.0%)
10/46 (21.7%)
3/14 (21.4%) / 38/187 (20.3%)
6/46 (13.0%)
3/14 (21.4%) / 0.576
Satisfaction with physician recommendations
No
Yes / 16/29 (55.2%)
147/229 (64.2%) / 8/29 (27.6%)
37/229 (16.2%) / 5/29 (17.2%)
45/229 (19.7%) / 0.311
Advocate to receive screening regimen
No
Yes / 98/153 (64.1%)
66/105 (62.9%) / 28/153 (18.3%)
17/105 (16.2%) / 27/153 (17.6%)
22/105 (21.0%) / 0.767
AFP frequency followed
6 weeks
3 months / 67/107 (62.6%)
64/101 (63.4%) / 20/107 (18.7%)
18/101 (17.8%) / 20/107 (18.7%)
19/101 (18.8%) / 0.987
AFP frequency recommended
6 weeks
3 months / 63/98 (64.3%)
73/119 (61.3%) / 19/98 (19.4%)
20/119 (16.8%) / 16/98 (16.3%)
26/119 (21.8%) / 0.571
AFP frequency beliefs
6 weeks
3 months / 91/140 (65.0%)
49/78 (62.8%) / 24/140 (17.1%)
13/78 (16.7%) / 25/140 (17.9%)
16/78 (20.5%) / 0.891
AFP frequency adherence
No
Yes / 134/207 (64.7%)
23/43 (53.5%) / 33/207 (15.9%)
10/43 (23.3%) / 40/207 (19.3%)
10/43 (23.3%) / 0.350
Knowledge
Incorrect
Correct / 45/66 (68.2%)
31/56 (55.4%) / 9/66 (13.6%)
13/56 (23.2%) / 12/66 (18.2%)
12/56 (21.4%) / 0.286
Parental Concern – Burden
Location Groups
United States
Other / 120/172 (69.8%)
51/82 (62.2%) / 26/172 (15.1%)
16/82 (19.5%) / 26/172 (15.1%)
15/82 (18.3%) / 0.480
Genetic Cause
IC2 LOM
Other / 48/76 (63.2%)
63/97 (64.9%) / 13/76 (17.1%)
17/97 (17.5%) / 15/76 (19.7%)
17/97 (17.5%) / 0.933
Number of Children
1 child
2 or more children / 28/48 (58.3%)
142/205 (69.3%) / 9/48 (18.8%)
33/205 (16.1%) / 11/48 (22.9%)
30/205 (14.6%) / 0.286
Child age groups
Under 4 years
4 - 7.99 years
8 years and over / 104/163 (63.8%)
44/58 (75.9%)
26/36 (72.2%) / 27/163 (16.6%)
8/58 (13.8%)
7/36 (19.4%) / 32/163 (19.6%)
6/58 (10.3%)
3/36 (8.3%) / 0.255
Employment Groups
Unemployed or Homemaker
Part-Time
Full-Time / 51/78 (65.4%)
30/40 (75.0%)
79/124 (63.7%) / 14/78 (17.9%)
7/40 (17.5%)
20/124 (16.1%) / 13/78 (16.7%)
3/40 (7.5%)
25/124 (20.2%) / 0.469
Martial Status
Living alone
Living with someone / 25/31 (80.6%)
146/223 (65.5%) / 4/31 (12.9%)
38/223 (17.0%) / 2/31 (6.5%)
39/223 (17.5%) / 0.195
Incurred Costs
No
Yes / 106/152 (69.7%)
67/104 (64.4%) / 28/152 (18.4%)
14/104 (13.5%) / 18/152 (11.8%)
23/104 (22.1%) / 0.072
History of false-positive screen
No
Yes / 117/174 (67.2%)
57/83 (68.7%) / 28/174 (16.1%)
14/83 (16.9%) / 29/174 (16.7%)
12/83 (14.5%) / 0.901
Satisfaction with physician recommendations
No
Yes / 19/29 (65.5%)
155/227 (68.3%) / 5/29 (17.2%)
37/227 (16.3%) / 5/29 (17.2%)
35/227 (15.4%) / 0.952
Advocate to receive screening regimen
No
Yes / 105/151 (69.5%)
69/105 (65.7%) / 23/151 (15.2%)
19/105 (18.1%) / 23/151 (15.2%)
17/105 (16.2%) / 0.787
AFP frequency followed
6 weeks
3 months / 71/106 (67.0%)
71/100 (71.0%) / 21/106 (19.8%)
14/100 (14.0%) / 14/106 (13.2%)
15/100 (15.0%) / 0.532
AFP frequency recommended
6 weeks
3 months / 67/97 (69.1%)
79/118 (66.9%) / 20/97 (20.6%)
18/118 (15.3%) / 10/97 (10.3%)
21/118 (17.8%) / 0.226
AFP frequency adherence
No
Yes / 142/205 (69.3%)
24/43 (55.8%) / 33/205 (16.1%)
9/43 (20.9%) / 30/205 (14.6%)
10/43 (23.3%) / 0.213
Parental Concern – Worry will Increase if Recommendations Change
Location Groups
United States
Other / 33/173 (19.1%)
13/83 (15.7%) / 41/173 (23.7%)
21/83 (25.3%) / 99/173 (57.2%)
49/83 (59.0%) / 0.797
Genetic Cause
IC2 LOM
Other / 18/77 (23.4%)
13/98 (13.3%) / 16/77 (20.8%)
25/98 (35.5%) / 43/77 (55.8%)
60/98 (61.2%) / 0.211
Number of Children
1 child
2 or more children / 9/49 (18.4%)
37/206 (18.0%) / 8/49 (16.3%)
54/206 (26.2%) / 32/49 (65.3%)
115/206 (55.8%) / 0.331
Child age groups
Under 4 years
4 - 7.99 years
8 years and over / 27/164 (16.5%)
13/58 (22.4%)
6/37 (16.2%) / 33/164 (20.1%)
17/58 (29.3%)
13/37 (35.1%) / 104/164 (63.4%)
28/58 (48.3%)
18/37 (48.6%) / 0.146
Education Groups
Associate’s degree or less
Bachelor’s degree
Advanced degree / 17/96 (17.7%)
13/83 (15.7%)
16/69 (23.2%) / 18/96 (18.8%)
22/83 (26.5%)
17/69 (24.6%) / 61/96 (63.5%)
48/83 (57.8%)
36/69 (52.2%) / 0.496
Employment Groups
Unemployed or Homemaker
Part-Time
Full-Time / 12/78 (15.4%)
6/41 (14.6%)
28/125 (22.4%) / 19/78 (24.4%)
15/41 (36.6%)
26/125 (20.8%) / 47/78 (60.3%)
20/41 (48.8%)
71/125 (56.8%) / 0.242
Martial Status
Living alone
Living with someone / 4/32 (12.5%)
42/224 (18.8%) / 6/32 (18.8%)
55/224 (24.6%) / 22/32 (68.8%)
127/224 (56.7%) / 0.427
Incurred Costs
No
Yes / 28/154 (18.2%)
18/104 (17.3%) / 34/154 (22.1%)
29/104 (27.9%) / 92/154 (59.7%)
57/104 (54.8%) / 0.564
History of false-positive screen
No
Yes / 25/176 (14.2%)
21/83 (25.3%) / 48/176 (27.3%)
15/83 (18.1%) / 103/176 (58.5%)
47/83 (56.6%) / 0.052
Risk threshold to screen
Any risk
1-5%
6-10% / 31/187 (16.6%)
8/46 (17.4%)
4/14 (28.6%) / 38/187 (20.3%)
16/46 (34.8%)
4/14 (28.6%) / 118/187 (63.1%)
22/46 (47.8%)
6/14 (42.9%) / 0.156
Satisfaction with physician recommendations
No
Yes / 4/29 (13.8%)
41/229 (17.9%) / 6/29 (20.7%)
57/229 (24.9%) / 19/29 (65.5%)
131/229 (57.2%) / 0.690
Advocate to receive screening
No
Yes / 32/153 (20.9%)
13/105 (12.4%) / 38/153 (24.8%)
25/105 (23.8%) / 83/153 (54.2%)
67/105 (63.8%) / 0.165
AFP frequency followed
6 weeks
3 months / 16/107 (15.0%)
17/101 (16.8%) / 25/107 (23.4%)
24/101 (23.8%) / 66/107 (61.7%)
60/101 (59.4%) / 0.922
AFP frequency recommended
6 weeks
3 months / 16/98 (16.3%)
20/119 (16.8%) / 22/98 (22.4%)
28/119 (23.5%) / 60/98 (61.2%)
71/119 (59.7%) / 0.972
AFP frequency beliefs
6 weeks
3 months / 19/140 (13.6%)
15/78 (19.2%) / 28/140 (20.0%)
21/78 (26.9%) / 93/140 (66.4%)
42/78 (53.8%) / 0.185
AFP frequency adherence
No
Yes / 39/207 (18.8%)
5/43 (11.6%) / 50/207 (24.2%)
9/43 (20.9%) / 118/207 (57.0%)
29/43 (67.4%) / 0.393
Knowledge
Incorrect
Correct / 12/66 (18.2%)
9/56 (16.1%) / 9/66 (13.6%)
17/56 (30.4%) / 45/66 (68.2%)
30/56 (53.6%) / 0.078