08-01-2019 | OriginalPaper | Uitgave 5/2019 Open Access

Toddler Screening for Autism Spectrum Disorder: A Meta-Analysis of Diagnostic Accuracy
- Tijdschrift:
- Journal of Autism and Developmental Disorders > Uitgave 5/2019
Electronic supplementary material
Publisher’s Note
Study number
|
Screening test(s)
|
Country
|
FN
a strategy
|
FN
|
FP
|
TP
|
TN
|
N
|
Total
N
b
|
Sex
|
Not reported
|
Age (months)
|
|
---|---|---|---|---|---|---|---|---|---|---|---|---|---|
Female
|
Male
|
||||||||||||
1. Nygren et al. (
2012)
|
M-CHAT
|
Sweden
|
No
|
NA
|
3
|
33
|
NA
|
3.985
|
3.999
|
2.087
|
1.912
|
NA
|
29.00
|
2. Nygren et al. (
2012)
|
JOBS
|
Sweden
|
No
|
NA
|
3
|
37
|
NA
|
3.985
|
3.999
|
2.087
|
1.912
|
NA
|
29.00
|
3. Nygren et al. (
2012)
|
M-CHAT + JOBS
|
Sweden
|
No
|
NA
|
5
|
43
|
NA
|
3.985
|
3.999
|
2.087
|
1.912
|
NA
|
29.00
|
4. Baird et al. (
2000)
|
CHAT
|
UK
|
Yes
|
74
|
14
|
20
|
16.127
|
16.235
|
NA
|
NA
|
NA
|
NA
|
18.70
|
5. Wiggins et al. (
2014)
|
M-CHAT
|
USA
|
Yes
|
3
|
17
|
27
|
3.803
|
3.850
|
3.980
|
NA
|
NA
|
NA
|
21.10
|
6. Wiggins et al. (
2014)
|
PEDS+ PATH
|
USA
|
Yes
|
2
|
20
|
28
|
2.978
|
3.028
|
3.980
|
NA
|
NA
|
NA
|
21.10
|
7. Kamio et al. (
2014)
|
M-CHAT_JV
|
Japan
|
Yes
|
22
|
24
|
20
|
1.661
|
1.727
|
2.141
|
880
|
847
|
NA
|
18.70
|
8. Stenberg et al. (
2014)
|
M-CHAT
|
Norway
|
Yes
|
114
|
3.804
|
59
|
48.049
|
52.026
|
NA
|
25.429
|
26.597
|
NA
|
18.00
|
9. Chlebowski et al. (
2013)
|
M-CHAT/Yale Screener + STAT
|
USA
|
Yes
|
6
|
79
|
92
|
18.269
|
18.446
|
18.989
|
9.388
|
9.601
|
NA
|
20.40
|
10. Canal-Bedia et al. (
2011)
|
M-CHAT
|
Spain
|
Yes
|
0
|
25
|
6
|
2.024
|
2.055
|
NA
|
949
|
1.106
|
NA
|
21.40
|
11. Barbaro and Dissanayake (
2010)
|
SACS
|
Australia
|
Yes
|
34
|
41
|
174
|
20.521
|
20.770
|
NA
|
10.177
|
10.593
|
NA
|
19.27
|
12. Inada et al. (
2011)
|
M-CHAT (short version 9, cut-off 1)
|
Japan
|
NA
|
NA
|
NA
|
20
|
NA
|
1.167
|
1.187
|
571
|
596
|
NA
|
18.00
|
13. Inada et al. (
2011)
|
M-CHAT (full version)
|
Japan
|
NA
|
NA
|
NA
|
20
|
NA
|
1.167
|
1.187
|
571
|
596
|
NA
|
18.00
|
14. Dereu et al. (
2010)
|
CESDD
|
Belgium
|
Yes
|
13
|
265
|
28
|
6.502
|
6.808
|
NA
|
3.255
|
3.553
|
NA
|
16.70
|
15. Miller et al. (
2011)
|
ITC + M-CHAT
|
USA
|
Yes
|
2
|
17
|
10
|
638
|
667
|
796
|
NA
|
NA
|
NA
|
NA
|
16. Robins et al. (
2014)
|
M-CHAT-R/F
|
USA
|
Yes
|
18
|
116
|
105
|
15.373
|
15.612
|
16.071
|
7.570
|
7.793
|
249
|
20.95
|
17. Honda et al. (
2005)
|
YACHT-18
|
Japan
|
Yes
|
16
|
NA
|
68
|
NA
|
35.716
|
NA
|
17.468
|
18.248
|
NA
|
18.00
|
18. Baranek (
2015)
|
M-CHAT
|
USA
|
Yes
|
3
|
32
|
5
|
534
|
574
|
NA
|
300
|
268
|
6
|
24.73
|
Methods
Criteria for Selection of Studies
Literature Search
Assessment of Methodological Quality
Data Extraction
Study
|
Se
|
(95% CI)
|
Sp
|
(95% CI)
|
PPV
|
(95% CI)
|
NPV
|
(95% CI)
|
LR+
|
(95% CI)
|
LR−
|
(95% CI)
|
---|---|---|---|---|---|---|---|---|---|---|---|---|
Nygren et al. (
2012)
|
0.767
|
(0.614–0.882)
|
NA
|
NA
|
0.917
|
(0.775–0.982)
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
Nygren et al. (
2012)
|
0.860
|
(0.721–0.947)
|
NA
|
NA
|
0.925
|
(0.796–0.984)
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
Nygren et al. (
2012)
|
0.956
|
(0.849–0.995)
|
NA
|
NA
|
0.896
|
(0.773–0.965)
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
Baird et al. (
2000)
|
0.213
|
(0.130–0.300)
|
0.999
|
(0.999–1.000)
|
0.588
|
(0.420–0.750)
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
Wiggins et al. (
2014)
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
Wiggins et al. (
2014)
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
Kamio et al. (
2014)
|
0.480
|
(0.330–0.630)
|
0.990
|
(0.980–0.990)
|
0.450
|
(0.310–0.600)
|
0.990
|
(0.980–0.990)
|
NA
|
NA
|
NA
|
NA
|
Stenberg et al. (
2014)
|
0.341
|
(0.271–0.417)
|
0.927
|
(0.924–0.929)
|
0.150
|
(0.120–0.200)
|
NA
|
NA
|
4.60
|
NA
|
NA
|
NA
|
Chlebowski et al. (
2013)
|
NA
|
NA
|
NA
|
NA
|
0.538
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
Canal-Bedia et al. (
2011)
|
1.000
|
NA
|
0.980
|
(0.980–0.990)
|
0.190
|
(0.050–0.330)
|
1.000
|
NA
|
NA
|
NA
|
NA
|
NA
|
Barbaro and Dissanayake (
2010)
|
0.836
|
(0.776–0.882)
|
0.998
|
(0.998–0.999)
|
0.807
|
(0.748–0.856)
|
0.998
|
(0.998–0.999)
|
414.39
|
(303.93–564.99)
|
0.17
|
(0.12–0.22)
|
Inada et al. (
2011)
|
0.650
|
NA
|
0.885
|
NA
|
0.088
|
NA
|
0.993
|
NA
|
NA
|
NA
|
NA
|
NA
|
Inada et al. (
2011)
|
0.550
|
NA
|
0.961
|
NA
|
0.193
|
NA
|
0.992
|
NA
|
NA
|
NA
|
NA
|
NA
|
Dereu et al. (
2010)
|
0.680
|
(0.540–0.830)
|
0.960
|
(0.960–0.970)
|
0,100
|
(0.060–0.130)
|
1.000
|
(0.999–1.00)
|
17.42
|
NA
|
0.33
|
NA
|
Miller et al. (
2011)
|
NA
|
NA
|
NA
|
NA
|
NA
|
NA
|
0.996
|
NA
|
NA
|
NA
|
NA
|
NA
|
Robins et al. (
2014)
|
0.854
|
NA
|
0.993
|
NA
|
0.475
|
NA
|
0.999
|
NA
|
114.05
|
NA
|
0.15
|
NA
|
Honda et al. (
2005)
|
0.810
|
NA
|
NA
|
NA
|
NA
|
NA
|
0.999
|
NA
|
NA
|
NA
|
NA
|
NA
|
Baranek (
2015)
|
0.625
|
(0.508–0.960)
|
0.943
|
NA
|
0.135
|
NA
|
0.994
|
NA
|
NA
|
NA
|
0.40
|
NA
|
Data Synthesis and Statistical Analysis
Results
Study Selection
Methodological Quality of the Included Studies
Characteristics of the Included Studies
Diagnostic Accuracy of Screening Tools
Parameters
|
Meta-analysis with all studies selected (N = 18)
|
Meta-analysis: subgroup of analysis (N = 9)
|
||||||||
---|---|---|---|---|---|---|---|---|---|---|
Estimated
|
SD
|
MC_error
|
C.I._lower
|
C.I._upper
|
Estimated
|
SD
|
MC_error
|
C.I._lower
|
C.I._upper
|
|
HSROC THETA
a
|
0.86
|
0.13
|
< 0.01
|
0.12
|
0.60
|
0.51
|
0.16
|
0.01
|
0.16
|
0.17
|
HSROC LAMBDA
b
|
2.89
|
0.13
|
< 0.01
|
2.59
|
2.99
|
2.90
|
0.14
|
< 0.01
|
2.56
|
2.99
|
HSROC Beta
c
|
− 0.09
|
< 0.01
|
< 0.01
|
− 0.09
|
− 0.09
|
0.38
|
0.09
|
0.01
|
0.20
|
0.55
|
σ
α
d
|
1.09
|
0.21
|
< 0.01
|
0.74
|
1.57
|
1.07
|
0.31
|
0.01
|
0.59
|
1.77
|
σ
θ
e
|
0.51
|
0.10
|
< 0.01
|
0.35
|
0.75
|
0.32
|
0.13
|
< 0.01
|
0.14
|
0.60
|
Se overall
|
0.72
|
0.05
|
< 0.01
|
0.61
|
0.81
|
0.77
|
0.03
|
< 0.01
|
0.69
|
0.84
|
Sp overall
|
0.98
|
< 0.01
|
< 0.01
|
0.97
|
0.99
|
0.99
|
< 0.01
|
< 0.01
|
0.97
|
0.99
|
Study
|
Screening test
|
THETA
a (95% CI)
|
ALPHA
b (95% CI)
|
Prevalence
c (95% CI)
|
Sensitivity (
Se) (95% CI)
|
Specificity (
Sp) (95% CI)
|
|||||
---|---|---|---|---|---|---|---|---|---|---|---|
Estimated
|
SD
|
Estimated
|
SD
|
Estimated
|
SD
|
Estimated
|
SD
|
Estimated
|
SD
|
||
Nygren et al. (
2012)
|
M-CHAT
|
1.31 (1.06–1.56)
|
0.12
|
3.95 (3.45–4.46)
|
0.24
|
0.01 (< 0.01–0.01)
|
< 0.01
|
0.75 (0.63–0.87)
|
0.06
|
0.99 (0.99–1)
|
< 0.01
|
Nygren et al. (
2012)
|
JOBS
|
1.16 (0.89–1.41)
|
0.13
|
4.21 (3.72–4.72)
|
0.25
|
0.01 (< 0.01–0.01)
|
< 0.01
|
0.84 (0.72–0.93)
|
0.05
|
0.99 (0.99–1)
|
< 0.01
|
Nygren et al. (
2012)
|
M-CHAT + JOBS
|
0.86 (0.58–1.12)
|
0.13
|
4.52 (4.02–5.03)
|
0.25
|
0.01 (< 0.01–0.01)
|
< 0.01
|
0.92 (0.85–0.98)
|
0.03
|
0.99 (0.99–1)
|
< 0.01
|
Baird et al. (
2000)
|
CHAT
|
1.99 (1.84–2.15)
|
0.07
|
2.58 (2.27–2.86)
|
0.15
|
< 0.01 (< 0.01 to < 0.01)
|
< 0.01
|
0.22 (0.15–0.31)
|
0.04
|
0.99 (0.99–1)
|
< 0.01
|
Wigginset al. (
2014)
|
M-CHAT
|
0.81 (0.53–1.05)
|
0.13
|
3.86 (3.37–4.40)
|
0.26
|
< 0.01 (< 0.01–0.01)
|
< 0.01
|
0.88 (0.77–0.96)
|
0.05
|
0.99 (0.99–1)
|
< 0.01
|
Wigginset al. (
2014)
|
PEDS + PATH
|
0.65 (0.39–0.94)
|
0.13
|
3.88 (3.33–4.44)
|
0.28
|
0.01 (< 0.01–0.01)
|
< 0.01
|
0.91 (0.80–0.97)
|
0.04
|
0.99 (0.99–1)
|
< 0.01
|
Kamio et al. (
2014)
|
M-CHAT_JV
|
1.15 (0.98–1.35)
|
0.09
|
2.28 (1.89–2.64)
|
0.19
|
0.02 (0.01–0.03)
|
< 0.01
|
0.49 (0.35–0.62)
|
0.07
|
0.98 (0.98–0.99)
|
< 0.01
|
Stenberg et al. (
2014)
|
M-CHAT
|
− 0.05 (− 0.14–0.01)
|
0.05
|
3.13 (2.97–3.31)
|
0.09
|
< 0.01 (< 0.01 to < 0.01)
|
< 0.01
|
0.95 (0.93–0.97)
|
< 0.01
|
0.92 (0.92–0.93)
|
< 0.01
|
Chlebowski et al. (
2013)
|
M-CHAT /YALE SCREENER and STAT
|
0.76 (0.59–0.91)
|
0.08
|
3.98 (3.68–4.30)
|
0.15
|
< 0.01 (< 0.01 to < 0.01)
|
< 0.01
|
0.90 (0.84–0.95)
|
0.02
|
0.99 (0.99–1)
|
< 0.01
|
Canal-Bedia et al. (
2011)
|
M-CHAT
|
0.54 (− 0.01 to − 1.03)
|
0.26
|
3.63 (2.63–4.69)
|
0.52
|
< 0.01 (< 0.01 to < 0.01)
|
< 0.01
|
0.90 (0.68–0.99)
|
0.09
|
0.98 (0.98–0.99)
|
< 0.01
|
Barbaro and Dissanayake (
2010)
|
SACS
|
1.06 (0.96–1.16)
|
0.05
|
3.90 (3.70–4.10)
|
0.10
|
0.01 (< 0.01–0.01)
|
< 0.01
|
0.82 (0.77–0.87)
|
0.02
|
0.99 (0.99–1)
|
< 0.01
|
Inada et al. (
2011)
|
M-CHAT (short version 9, cutoff:1)
|
0.23 (< 0.01–0.43)
|
0.10
|
1.44 (1.02–1.85)
|
0.20
|
0.02 (0.01–0.03)
|
< 0.01
|
0.69 (0.54–0.83)
|
0.07
|
0.81 (0.79–0.84)
|
0.01
|
Inada et al. (
2011)
|
M-CHAT (full version)
|
0.66 (0. 47–0.84)
|
0.09
|
1.71 (1.31–2.07)
|
0.19
|
0.03 (0.02–0.04)
|
< 0.01
|
0.58 (0.43–0.72)
|
0.07
|
0.92 (0.91–0.94)
|
< 0.01
|
Dereu et al. (
2010)
|
CESDD
|
0.68 (0.56–0.83)
|
0.07
|
2.32 (2.02–2.59)
|
0.15
|
< 0.01 (< 0.01 to <0.01)
|
< 0.01
|
0.69 (0.58–0.77)
|
0.05
|
0.96 (0.95–0.96)
|
< 0.01
|
Miller et al. (
2011)
|
ITC + M-CHAT
|
0.61 (0.27–0.93)
|
0.17
|
2.89 (2.23–3.61)
|
0.34
|
0.01 (0.01–0.03)
|
< 0.01
|
0.81 (0.62–0.96)
|
0.08
|
0.97 (0.96–0.98)
|
< 0.01
|
Robins et al. (
2014)
|
M-CHAT-R/F
|
0.78 (0.67–0.91)
|
0.06
|
3.53 (3.27–3.79)
|
0.13
|
< 0.01 (< 0.01 to < 0.01)
|
< 0.01
|
0.84 (0.78–0.90)
|
0.03
|
0.99 (0.99–1)
|
< 0.01
|
Honda et al. (
2005)
|
YACHT-18
|
1.58 (1.41–1.75)
|
0.08
|
4.27 (4.00–4.56)
|
0.14
|
< 0.01 (< 0.01–<0.01)
|
< 0.01
|
0.71 (0.63–0.79)
|
0.04
|
0.99 (0.99–1)
|
< 0.01
|
Baranek (
2015)
|
M-CHAT
|
0.68 (0.31–1.33)
|
0.18
|
1.99 (1.27–2.71)
|
0.37
|
0.01 (< 0.01–0.01)
|
< 0.01
|
0.62 (0.35–0.85)
|
0.13
|
0.94 (0.92–0.96)
|
< 0.01
|
Exploration of Heterogeneity
Subgroup of Analysis
Study
|
Screening test
|
THETA
a (95% CI)
|
ALPHA
b (95% CI)
|
Prevalence
c (95% CI)
|
Se (95% CI)
|
Sp (95% CI)
|
|||||
---|---|---|---|---|---|---|---|---|---|---|---|
Estimated
|
SD
|
Estimated
|
SD
|
Estimated
|
SD
|
Estimated
|
SD
|
Estimated
|
SD
|
||
Nygren et al. (
2012)
|
M-CHAT
|
0.82 (0.47–1.14)
|
0.17
|
3.56 (3.45–4.46)
|
0.29
|
0.01 (< 0.01–0.01)
|
< 0.01
|
0.78 (0.65–0.90)
|
0.06
|
0.99 (0.99–1)
|
< 0.01
|
Nygren et al. (
2012)
|
JOBS
|
0.65 (0.31–0.98)
|
0.17
|
3.93 (3.72–4.72)
|
0.28
|
0.01 (< 0.01 -01)
|
< 0.01
|
0.86 (0.76–0.94)
|
0.05
|
0.99 (0.99–1)
|
< 0.01
|
Nygren et al. (
2012)
|
M-CHAT + JOBS
|
0.34 (-0.03–0.71)
|
0.19
|
4.32 (4.02–5.03)
|
0.33
|
0.01 (< 0.01–0.01)
|
< 0.01
|
0.93 (0.85–0.98)
|
0.03
|
0.99 (0.99–1)
|
< 0.01
|
Wiggins et al. (
2014)
|
M-CHAT
|
0.35 (− 0.06 to 0.76)
|
0.20
|
3.61 (3.37–4.40)
|
0.33
|
< 0.01 (< 0.01–0.01)
|
< 0.01
|
0.88 (0.76–0.96)
|
0.05
|
0.99 (0.99–1)
|
< 0.01
|
Wiggins et al. (
2014)
|
PEDS + PATH
|
0.24 (− 0.15 to 0.76)
|
0.20
|
3.57 (3.33–4.44)
|
0.36
|
0.01 (< 0.01–0.01)
|
< 0.01
|
0.89 (0.77–0.98)
|
0.04
|
0.99 (0.99–1)
|
< 0.01
|
Chlebowski et al. (
2013)
|
M-CHAT /YALE SCREENER/STAT
|
0.24 (0.04–0.42)
|
0.10
|
3.87 (3.68–4.30)
|
0.21
|
< 0.01 (< 0.01 to < 0.01)
|
< 0.01
|
0.91 (0.85–0.95)
|
0.02
|
0.99 (0.99–1)
|
< 0.01
|
Barbaro and Dissanayake (
2010)
|
SACS
|
0.60 (0.36–0.81)
|
0.10
|
3.56 (3.70–4.10)
|
0.14
|
0.01 (< 0.01 to < 0.01)
|
< 0.01
|
0.83 (0.78–0.88)
|
0.02
|
0.99 (0.99–1)
|
< 0.01
|
Robins et al. (
2014)
|
M-CHAT-R/F
|
0.36 (0.14–0.49)
|
0.08
|
3.26 (3.27–3.79)
|
0.15
|
< 0.01 (< 0.01 to <0.01)
|
< 0.01
|
0.85 (0.80–0.91)
|
0.03
|
0.99 (0.99–1)
|
< 0.01
|
Honda et al. (
2005)
|
YACHT-18
|
0.98 (0.66–1.29)
|
0.16
|
4.15 (4.00–4.56)
|
0.20
|
< 0.01 (< 0.01 to <0.01)
|
< 0.01
|
0.81 (0.73–0.89)
|
0.04
|
0.99 (0.99–1)
|
< 0.01
|