1 meta-analysis with missing data: metamiss ian white and julian higgins mrc biostatistics unit,...

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1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September 2007

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Page 1: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

1

Meta-analysis with missing data: metamiss

Ian White and Julian HigginsMRC Biostatistics Unit, Cambridge, UK

Stata users’ group, London

10 September 2007

Page 2: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

2

Motivation

• Missing outcome data compromise trials

• So they also compromise meta-analyses

• We may want to – correct for bias due to missing data– down-weight trials with more missing data

• NB missing data within trials, not missing trials

Page 3: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

3

Plan

Meta-analysis of binary data

• Haloperidol example

• Standard approaches to missing data

• Imputation methods

• IMORs

• Methods that allow for uncertainty

• Demonstration

Page 4: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

4

Haloperidol meta-analysis  Haloperidol Placebo

% missing   r1 f1 m1 n1 r2 f2 m2 n2

Arvanitis 25 25 2 52 18 33 0 51 2%

Beasley 29 18 22 69 20 14 34 68 41%

Bechelli 12 17 1 30 2 28 1 31 3%

Borison 3 9 0 12 0 12 0 12 0%

Chouinard 10 11 0 21 3 19 0 22 0%

Durost 11 8 0 19 1 14 0 15 0%

Garry 7 18 1 26 4 21 1 26 4%

Howard 8 9 0 17 3 10 0 13 0%

Marder 19 45 2 66 14 50 2 66 3%

Nishikawa 82 1 9 0 10 0 10 0 10 0%

Nishikawa 84 11 23 3 37 0 13 0 13 6%

Reschke 20 9 0 29 2 9 0 11 0%

Selman 17 1 11 29 7 4 18 29 50%

Serafetinides 4 10 0 14 0 13 1 14 4%

Simpson 2 14 0 16 0 7 1 8 4%

Spencer 11 1 0 12 1 11 0 12 0%

Vichaiya 9 20 1 30 0 29 1 30 3%

r=successes

f=failures

m=missing

n=total

Page 5: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

5

Standard approaches to missing data

• Available cases (complete cases): ignore the missing data– assumes MAR: missingness is independent of

outcome given arm

• Assume missing=failure– implausible, but not too bad for health-related

behaviours

• Neither assumption is likely to be correct

Page 6: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

6

Other ideas• Sensitivity analyses, e.g. do both

missing=failure and available cases– but these could agree by chance

• Explore best / worst cases• Use reasons for missingness• Explicit assumptions about informative

missingness (IM)– IM: missingness is dependent on outcome

Page 7: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

7

metamiss.ado• Processes data on successes, failures and missing by arm

& feeds results to metan• Available cases analysis (ACA)• Imputed case analyses (ICA):

– impute as failure: ICA-0– impute as success: ICA-1– best-case: ICA-b (missing=success in E, failure in C)– worst-case: ICA-w– impute with same probability as in control arm: ICA-pC– impute with same probability as in experimental arm: ICA-pE– impute with same probability as in own arm: ICA-p (agrees with

ACA)– impute using IMORs: ICA-IMOR (see next slide)

Page 8: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

8

More general imputation: IMORs• Measure Informative Missingness using the

Informative Missing Odds Ratio (IMOR):– Odds ratio between outcome and missingness

• Can’t estimate IMOR from the data, but given any value of IMOR, we can analyse the data

• Generalises other ideas: e.g. – ICA-0 uses IMORs 0, 0– ICA-1 uses IMORs , – ICA-b uses IMORs , 0– ICA-p uses IMORs 1, 1– ICA-pC uses IMORs OR, 1 where OR is odds ratio

between arm and outcome in available cases

Page 9: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

9

Getting standard errors (weighting) right

• Weight 1: treat imputed data as real• Weight 2: use standard errors from ACA• Weight 3: scale imputed data to same sample size

as available cases• Weight 4: algebraic standard errors

– same as weight 1 for ICA-0, ICA-1, ICA-b, ICA-w– same as weight 2 for ICA-p– uses Taylor expansion for ICA-IMOR– for ICA-pC & ICA-pE, we condition on the IMOR (I

can explain…)

Page 10: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

10

Page 11: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

11

Page 12: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

12

Overall (I-squared = 41.4%, p = 0.038)

Arvantis

Vichaiya

Selman

Chouinard

Nishikawa_84

Howard

Serafetinides

Durost

Simpson

Mander

Reschke

Study ID

Garry

Borison_92

Spencer

Beasley

Bechelli

Nishikawa_82

1.57 (1.28, 1.92)

1.42 (0.89, 2.25)

19.00 (1.16, 311.96)

1.48 (0.94, 2.35)

3.49 (1.11, 10.95)

9.20 (0.58, 145.76)

2.04 (0.67, 6.21)

8.40 (0.50, 142.27)

8.68 (1.26, 59.95)

2.35 (0.13, 43.53)

1.36 (0.75, 2.47)

3.79 (1.06, 13.60)

ES (95% CI)

1.75 (0.58, 5.24)

7.00 (0.40, 122.44)

11.00 (1.67, 72.40)

1.05 (0.73, 1.50)

6.21 (1.52, 25.35)

3.00 (0.14, 65.90)

100.00

18.86

0.52

19.11

3.10

0.53

3.27

0.51

1.09

0.48

11.37

2.48

Weight

3.37

%

0.49

1.14

31.22

2.05

0.42

1.57 (1.28, 1.92)

1.42 (0.89, 2.25)

19.00 (1.16, 311.96)

1.48 (0.94, 2.35)

3.49 (1.11, 10.95)

9.20 (0.58, 145.76)

2.04 (0.67, 6.21)

8.40 (0.50, 142.27)

8.68 (1.26, 59.95)

2.35 (0.13, 43.53)

1.36 (0.75, 2.47)

3.79 (1.06, 13.60)

ES (95% CI)

1.75 (0.58, 5.24)

7.00 (0.40, 122.44)

11.00 (1.67, 72.40)

1.05 (0.73, 1.50)

6.21 (1.52, 25.35)

3.00 (0.14, 65.90)

100.00

18.86

0.52

19.11

3.10

0.53

3.27

0.51

1.09

0.48

11.37

2.48

Weight

3.37

%

0.49

1.14

31.22

2.05

0.42

1.1 1 10 100

ACA

Page 13: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

13

Overall (I-squared = 25.8%, p = 0.158)

Study ID

Borison_92

Chouinard

Durost

Vichaiya

Selman

Beasley

Nishikawa_82

Howard

Simpson

Nishikawa_84

Mander

Spencer

Garry

Arvantis

Reschke

Bechelli

Serafetinides

1.90 (1.51, 2.39)

ES (95% CI)

7.00 (0.40, 122.44)

3.49 (1.11, 10.95)

8.68 (1.26, 59.95)

19.00 (1.16, 312.42)

2.43 (1.19, 4.96)

1.43 (0.90, 2.27)

3.00 (0.14, 65.90)

2.04 (0.67, 6.21)

2.65 (0.14, 49.42)

8.47 (0.53, 134.46)

1.36 (0.74, 2.47)

11.00 (1.67, 72.40)

1.75 (0.58, 5.27)

1.36 (0.85, 2.17)

3.79 (1.06, 13.60)

6.20 (1.51, 25.40)

9.00 (0.53, 152.93)

100.00

Weight

0.65

4.06

1.42

0.68

10.42

%

25.01

0.56

4.29

0.62

0.70

14.75

1.50

4.38

24.38

3.26

2.67

0.66

1.90 (1.51, 2.39)

ES (95% CI)

7.00 (0.40, 122.44)

3.49 (1.11, 10.95)

8.68 (1.26, 59.95)

19.00 (1.16, 312.42)

2.43 (1.19, 4.96)

1.43 (0.90, 2.27)

3.00 (0.14, 65.90)

2.04 (0.67, 6.21)

2.65 (0.14, 49.42)

8.47 (0.53, 134.46)

1.36 (0.74, 2.47)

11.00 (1.67, 72.40)

1.75 (0.58, 5.27)

1.36 (0.85, 2.17)

3.79 (1.06, 13.60)

6.20 (1.51, 25.40)

9.00 (0.53, 152.93)

100.00

Weight

0.65

4.06

1.42

0.68

10.42

%

25.01

0.56

4.29

0.62

0.70

14.75

1.50

4.38

24.38

3.26

2.67

0.66

1.1 1 10 100

ICA-0

Page 14: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

14

Overall (I-squared = 60.3%, p = 0.001)

Spencer

Study ID

Beasley

Serafetinides

Borison_92

Garry

Selman

Mander

Nishikawa_82

Durost

Howard

Chouinard

Nishikawa_84

Bechelli

Arvantis

Reschke

Vichaiya

Simpson

1.16 (1.04, 1.29)

11.00 (1.67, 72.40)

ES (95% CI)

0.93 (0.77, 1.12)

4.00 (0.51, 31.46)

7.00 (0.40, 122.44)

1.60 (0.60, 4.25)

1.12 (0.95, 1.32)

1.31 (0.75, 2.28)

3.00 (0.14, 65.90)

8.68 (1.26, 59.95)

2.04 (0.67, 6.21)

3.49 (1.11, 10.95)

10.68 (0.68, 167.43)

4.48 (1.42, 14.15)

1.47 (0.93, 2.32)

3.79 (1.06, 13.60)

10.00 (1.36, 73.33)

1.00 (0.11, 9.44)

100.00

0.35

Weight

35.81

0.29

0.15

1.29

47.38

4.01

0.13

0.33

0.99

0.94

0.16

0.93

5.95

0.75

0.31

0.24

%

1.16 (1.04, 1.29)

11.00 (1.67, 72.40)

ES (95% CI)

0.93 (0.77, 1.12)

4.00 (0.51, 31.46)

7.00 (0.40, 122.44)

1.60 (0.60, 4.25)

1.12 (0.95, 1.32)

1.31 (0.75, 2.28)

3.00 (0.14, 65.90)

8.68 (1.26, 59.95)

2.04 (0.67, 6.21)

3.49 (1.11, 10.95)

10.68 (0.68, 167.43)

4.48 (1.42, 14.15)

1.47 (0.93, 2.32)

3.79 (1.06, 13.60)

10.00 (1.36, 73.33)

1.00 (0.11, 9.44)

100.00

0.35

Weight

35.81

0.29

0.15

1.29

47.38

4.01

0.13

0.33

0.99

0.94

0.16

0.93

5.95

0.75

0.31

0.24

%

1.1 1 10 100

ICA-1

Page 15: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

15

Overall (I-squared = 25.9%, p = 0.156)

Study ID

Bechelli

Chouinard

Borison_92

Simpson

Nishikawa_84

Mander

Beasley

Nishikawa_82

Serafetinides

Reschke

Durost

Howard

Selman

Spencer

Arvantis

Garry

Vichaiya

2.42 (1.95, 3.00)

ES (95% CI)

6.72 (1.65, 27.28)

3.49 (1.11, 10.95)

7.00 (0.40, 122.44)

2.65 (0.14, 49.42)

10.68 (0.68, 167.43)

1.50 (0.84, 2.69)

2.51 (1.69, 3.73)

3.00 (0.14, 65.90)

9.00 (0.53, 152.93)

3.79 (1.06, 13.60)

8.68 (1.26, 59.95)

2.04 (0.67, 6.21)

4.00 (2.09, 7.65)

11.00 (1.67, 72.40)

1.47 (0.93, 2.32)

2.00 (0.69, 5.83)

21.00 (1.29, 342.93)

100.00

Weight

2.37

3.57

0.57

0.54

0.62

13.68

30.05

%

0.49

0.58

2.86

1.25

3.76

11.08

1.31

22.59

4.07

0.60

2.42 (1.95, 3.00)

ES (95% CI)

6.72 (1.65, 27.28)

3.49 (1.11, 10.95)

7.00 (0.40, 122.44)

2.65 (0.14, 49.42)

10.68 (0.68, 167.43)

1.50 (0.84, 2.69)

2.51 (1.69, 3.73)

3.00 (0.14, 65.90)

9.00 (0.53, 152.93)

3.79 (1.06, 13.60)

8.68 (1.26, 59.95)

2.04 (0.67, 6.21)

4.00 (2.09, 7.65)

11.00 (1.67, 72.40)

1.47 (0.93, 2.32)

2.00 (0.69, 5.83)

21.00 (1.29, 342.93)

100.00

Weight

2.37

3.57

0.57

0.54

0.62

13.68

30.05

%

0.49

0.58

2.86

1.25

3.76

11.08

1.31

22.59

4.07

0.60

1.1 1 10 100

ICA-B

Page 16: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

16

Overall (I-squared = 74.2%, p = 0.000)

Serafetinides

Arvantis

Durost

Beasley

Study ID

Simpson

Reschke

Selman

Borison_92

Mander

Nishikawa_82

Vichaiya

Chouinard

Bechelli

Nishikawa_84

Spencer

Garry

Howard

0.94 (0.79, 1.12)

4.00 (0.51, 31.46)

1.36 (0.85, 2.17)

8.68 (1.26, 59.95)

0.53 (0.39, 0.72)

ES (95% CI)

1.00 (0.11, 9.44)

3.79 (1.06, 13.60)

0.68 (0.48, 0.95)

7.00 (0.40, 122.44)

1.19 (0.67, 2.10)

3.00 (0.14, 65.90)

9.00 (1.21, 66.70)

3.49 (1.11, 10.95)

4.13 (1.29, 13.20)

8.47 (0.53, 134.46)

11.00 (1.67, 72.40)

1.40 (0.51, 3.85)

2.04 (0.67, 6.21)

100.00

0.72

13.99

0.82

33.33

Weight

0.60

%

1.87

26.57

0.37

9.35

0.32

0.76

2.33

2.26

0.40

0.86

2.98

2.46

0.94 (0.79, 1.12)

4.00 (0.51, 31.46)

1.36 (0.85, 2.17)

8.68 (1.26, 59.95)

0.53 (0.39, 0.72)

ES (95% CI)

1.00 (0.11, 9.44)

3.79 (1.06, 13.60)

0.68 (0.48, 0.95)

7.00 (0.40, 122.44)

1.19 (0.67, 2.10)

3.00 (0.14, 65.90)

9.00 (1.21, 66.70)

3.49 (1.11, 10.95)

4.13 (1.29, 13.20)

8.47 (0.53, 134.46)

11.00 (1.67, 72.40)

1.40 (0.51, 3.85)

2.04 (0.67, 6.21)

100.00

0.72

13.99

0.82

33.33

Weight

0.60

%

1.87

26.57

0.37

9.35

0.32

0.76

2.33

2.26

0.40

0.86

2.98

2.46

1.1 1 10 100

ICA-W

Page 17: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

17

Overall (I-squared = 42.0%, p = 0.035)

Spencer

Nishikawa_82

Study ID

Simpson

Serafetinides

Chouinard

Bechelli

Borison_92

Mander

Arvantis

Vichaiya

Selman

Reschke

Nishikawa_84

Garry

Beasley

Durost

Howard

1.53 (1.24, 1.88)

11.00 (1.67, 72.40)

3.00 (0.14, 65.90)

ES (95% CI)

2.35 (0.13, 43.53)

8.40 (0.50, 142.27)

3.49 (1.11, 10.95)

6.03 (1.47, 24.68)

7.00 (0.40, 122.44)

1.35 (0.74, 2.45)

1.40 (0.88, 2.23)

18.42 (1.12, 302.65)

1.30 (0.76, 2.23)

3.79 (1.06, 13.60)

8.55 (0.54, 135.71)

1.72 (0.57, 5.16)

1.03 (0.72, 1.49)

8.68 (1.26, 59.95)

2.04 (0.67, 6.21)

100.00

1.21

0.45

Weight

0.51

0.54

3.29

2.17

0.53

%

12.05

19.89

0.55

14.85

2.64

0.56

3.57

32.56

1.15

3.47

1.53 (1.24, 1.88)

11.00 (1.67, 72.40)

3.00 (0.14, 65.90)

ES (95% CI)

2.35 (0.13, 43.53)

8.40 (0.50, 142.27)

3.49 (1.11, 10.95)

6.03 (1.47, 24.68)

7.00 (0.40, 122.44)

1.35 (0.74, 2.45)

1.40 (0.88, 2.23)

18.42 (1.12, 302.65)

1.30 (0.76, 2.23)

3.79 (1.06, 13.60)

8.55 (0.54, 135.71)

1.72 (0.57, 5.16)

1.03 (0.72, 1.49)

8.68 (1.26, 59.95)

2.04 (0.67, 6.21)

100.00

1.21

0.45

Weight

0.51

0.54

3.29

2.17

0.53

%

12.05

19.89

0.55

14.85

2.64

0.56

3.57

32.56

1.15

3.47

1.1 1 10 100

ICA-pc

Page 18: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

18

Overall (I-squared = 41.4%, p = 0.038)

Garry

Vichaiya

Howard

Borison_92

Selman

Durost

Nishikawa_82

Reschke

Mander

Chouinard

Simpson

Spencer

Nishikawa_84

Bechelli

Serafetinides

Study ID

Beasley

Arvantis

1.57 (1.28, 1.92)

1.75 (0.58, 5.24)

19.00 (1.16, 311.96)

2.04 (0.67, 6.21)

7.00 (0.40, 122.44)

1.48 (0.94, 2.35)

8.68 (1.26, 59.95)

3.00 (0.14, 65.90)

3.79 (1.06, 13.60)

1.36 (0.75, 2.47)

3.49 (1.11, 10.95)

2.35 (0.13, 43.53)

11.00 (1.67, 72.40)

9.20 (0.58, 145.76)

6.21 (1.52, 25.35)

8.40 (0.50, 142.27)

ES (95% CI)

1.05 (0.73, 1.50)

1.42 (0.89, 2.25)

100.00

3.37

0.52

3.27

0.49

19.11

1.09

0.42

2.48

11.37

3.10

0.48

1.14

0.53

2.05

0.51

Weight

31.22

18.86

%

1.57 (1.28, 1.92)

1.75 (0.58, 5.24)

19.00 (1.16, 311.96)

2.04 (0.67, 6.21)

7.00 (0.40, 122.44)

1.48 (0.94, 2.35)

8.68 (1.26, 59.95)

3.00 (0.14, 65.90)

3.79 (1.06, 13.60)

1.36 (0.75, 2.47)

3.49 (1.11, 10.95)

2.35 (0.13, 43.53)

11.00 (1.67, 72.40)

9.20 (0.58, 145.76)

6.21 (1.52, 25.35)

8.40 (0.50, 142.27)

ES (95% CI)

1.05 (0.73, 1.50)

1.42 (0.89, 2.25)

100.00

3.37

0.52

3.27

0.49

19.11

1.09

0.42

2.48

11.37

3.10

0.48

1.14

0.53

2.05

0.51

Weight

31.22

18.86

%

1.1 1 10 100

ICA-p

Page 19: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

19

Overall (I-squared = 48.2%, p = 0.014)

Vichaiya

Nishikawa_82

Garry

Bechelli

Simpson

Beasley

Howard

Selman

Borison_92

Nishikawa_84

Reschke

Mander

Durost

Chouinard

Spencer

Study ID

Serafetinides

Arvantis

1.42 (1.19, 1.69)

18.73 (1.14, 306.64)

3.00 (0.14, 65.90)

1.74 (0.59, 5.16)

6.12 (1.51, 24.87)

2.14 (0.12, 38.74)

1.00 (0.74, 1.35)

2.04 (0.67, 6.21)

1.32 (0.93, 1.86)

7.00 (0.40, 122.44)

9.57 (0.61, 151.28)

3.79 (1.06, 13.60)

1.35 (0.75, 2.45)

8.68 (1.26, 59.95)

3.49 (1.11, 10.95)

11.00 (1.67, 72.40)

ES (95% CI)

7.91 (0.47, 132.79)

1.43 (0.90, 2.28)

100.00

0.40

0.33

2.64

1.59

0.37

35.19

2.52

26.22

0.38

0.41

%

1.91

8.89

0.84

2.39

0.88

Weight

0.39

14.67

1.42 (1.19, 1.69)

18.73 (1.14, 306.64)

3.00 (0.14, 65.90)

1.74 (0.59, 5.16)

6.12 (1.51, 24.87)

2.14 (0.12, 38.74)

1.00 (0.74, 1.35)

2.04 (0.67, 6.21)

1.32 (0.93, 1.86)

7.00 (0.40, 122.44)

9.57 (0.61, 151.28)

3.79 (1.06, 13.60)

1.35 (0.75, 2.45)

8.68 (1.26, 59.95)

3.49 (1.11, 10.95)

11.00 (1.67, 72.40)

ES (95% CI)

7.91 (0.47, 132.79)

1.43 (0.90, 2.28)

100.00

0.40

0.33

2.64

1.59

0.37

35.19

2.52

26.22

0.38

0.41

%

1.91

8.89

0.84

2.39

0.88

Weight

0.39

14.67

1.1 1 10 100

ICA-IMOR 2 2

Page 20: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

20

Overall (I-squared = 35.0%, p = 0.077)

Spencer

Durost

Simpson

Study ID

Beasley

Nishikawa_82

Reschke

Bechelli

Garry

Howard

Serafetinides

Selman

Vichaiya

Mander

Borison_92

Arvantis

Chouinard

Nishikawa_84

1.70 (1.37, 2.11)

11.00 (1.67, 72.40)

8.68 (1.26, 59.95)

2.49 (0.13, 46.31)

ES (95% CI)

1.12 (0.74, 1.70)

3.00 (0.14, 65.90)

3.79 (1.06, 13.60)

6.23 (1.52, 25.49)

1.75 (0.58, 5.26)

2.04 (0.67, 6.21)

8.68 (0.51, 147.44)

1.74 (0.97, 3.12)

19.06 (1.16, 313.20)

1.36 (0.75, 2.47)

7.00 (0.40, 122.44)

1.40 (0.88, 2.23)

3.49 (1.11, 10.95)

8.91 (0.56, 141.28)

100.00

1.35

1.28

0.56

Weight

27.47

0.50

2.94

2.41

3.96

3.87

%

0.60

14.11

0.61

13.34

0.58

22.12

3.66

0.63

1.70 (1.37, 2.11)

11.00 (1.67, 72.40)

8.68 (1.26, 59.95)

2.49 (0.13, 46.31)

ES (95% CI)

1.12 (0.74, 1.70)

3.00 (0.14, 65.90)

3.79 (1.06, 13.60)

6.23 (1.52, 25.49)

1.75 (0.58, 5.26)

2.04 (0.67, 6.21)

8.68 (0.51, 147.44)

1.74 (0.97, 3.12)

19.06 (1.16, 313.20)

1.36 (0.75, 2.47)

7.00 (0.40, 122.44)

1.40 (0.88, 2.23)

3.49 (1.11, 10.95)

8.91 (0.56, 141.28)

100.00

1.35

1.28

0.56

Weight

27.47

0.50

2.94

2.41

3.96

3.87

%

0.60

14.11

0.61

13.34

0.58

22.12

3.66

0.63

1.1 1 10 100

ICA-IMOR 1/2 1/2

Page 21: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

21

Allowing for reasons (ICA-R)

• Specify number of missing individuals in each arm to be imputed by each scheme ICA-0, ICA-1, ICA-pC, ICA-pE, ICA-p, ICA-IMOR.

• Can take these data from a different outcome: metamiss scales to #missing

• If missing in a particular study, metamiss imputes using combined studies

Page 22: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

22

Page 23: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

23

Allowing for uncertainty

• So far we have pretended we really know the IMORs

• This is never really correct

• Now we allow them to be unknown but from a user-specified distribution

Page 24: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

24

Bayesian approach allowing for uncertain IMORs (Rubin, 1977)

E C

2E E E E

2E

E

E

Prior for , = log(IMOR) in experimental, control arm:

N ,

, measure your best guess about IM;

, measure your uncertainty about IM;

measu

C

C C C C

C

C

E Cres how similar you think , are:

0 is most conservative, 1 often allows little impact of IM on results.

Page 25: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

25

Bayesian analysis

• Elicit prior for E, C or use N(0,12) or N(0,22)• Get posterior distribution by integrating over the 2-

dimensional distribution of E, C. • metamiss does this fast & accurately by:

1. Standard normal approximation to posterior given E, C 2. Integrate using Gauss-Hermite quadrature.

• Alternatives: – Taylor expansion (inaccurate for large SD of log IMOR)– Full Bayesian Monte Carlo (slow, little gain in accuracy)

Page 26: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

26

Understanding priors for log IMOR: implied prior for P(success | missing)

when P(success | observed) = 1/2D

ensi

ty

0 .25 .5 .75 1P(success | missing)

N(0,0.5^2) N(0,2^2) N(-1,0.5^2) N(-1,2^2)

Page 27: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

27

Page 28: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

28

Overall (I-squared = 31.4%, p = 0.106)

Study ID

Borison_92

Simpson

Chouinard

Mander

Bechelli

Selman

Serafetinides

Spencer

Nishikawa_84

Garry

Durost

Nishikawa_82

Beasley

Reschke

Arvantis

Vichaiya

Howard

1.75 (1.38, 2.22)

ES (95% CI)

7.00 (0.40, 122.44)

2.27 (0.12, 42.39)

3.49 (1.11, 10.95)

1.35 (0.74, 2.47)

6.14 (1.50, 25.13)

1.54 (0.82, 2.88)

8.17 (0.48, 139.06)

11.00 (1.67, 72.40)

9.25 (0.58, 146.78)

1.74 (0.58, 5.23)

8.68 (1.26, 59.95)

3.00 (0.14, 65.90)

1.06 (0.61, 1.85)

3.79 (1.06, 13.60)

1.42 (0.89, 2.25)

18.74 (1.14, 308.29)

2.04 (0.67, 6.21)

100.00

Weight

%

0.68

0.65

4.26

15.46

2.80

14.02

0.69

1.57

0.73

4.60

1.49

0.58

18.07

3.41

25.78

0.71

4.49

1.75 (1.38, 2.22)

ES (95% CI)

7.00 (0.40, 122.44)

2.27 (0.12, 42.39)

3.49 (1.11, 10.95)

1.35 (0.74, 2.47)

6.14 (1.50, 25.13)

1.54 (0.82, 2.88)

8.17 (0.48, 139.06)

11.00 (1.67, 72.40)

9.25 (0.58, 146.78)

1.74 (0.58, 5.23)

8.68 (1.26, 59.95)

3.00 (0.14, 65.90)

1.06 (0.61, 1.85)

3.79 (1.06, 13.60)

1.42 (0.89, 2.25)

18.74 (1.14, 308.29)

2.04 (0.67, 6.21)

100.00

Weight

%

0.68

0.65

4.26

15.46

2.80

14.02

0.69

1.57

0.73

4.60

1.49

0.58

18.07

3.41

25.78

0.71

4.49

1.1 1 10 100

logimor ~ N(0,1)

Page 29: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

29

Overall (I-squared = 23.6%, p = 0.181)

Serafetinides

Vichaiya

Study ID

Nishikawa_84

Selman

Durost

Nishikawa_82

Arvantis

Bechelli

Simpson

Garry

Howard

Borison_92

Reschke

Mander

Chouinard

Beasley

Spencer

1.87 (1.44, 2.41)

7.61 (0.43, 133.33)

17.81 (1.06, 298.38)

ES (95% CI)

9.32 (0.59, 148.16)

1.60 (0.67, 3.80)

8.68 (1.26, 59.95)

3.00 (0.14, 65.90)

1.42 (0.89, 2.26)

5.98 (1.45, 24.71)

2.10 (0.11, 40.70)

1.73 (0.57, 5.22)

2.04 (0.67, 6.21)

7.00 (0.40, 122.44)

3.79 (1.06, 13.60)

1.35 (0.74, 2.47)

3.49 (1.11, 10.95)

1.08 (0.51, 2.32)

11.00 (1.67, 72.40)

100.00

0.80

0.83

Weight

%

0.86

8.77

1.77

0.69

30.37

3.28

0.75

5.40

5.32

0.81

4.04

18.04

5.05

11.36

1.86

1.87 (1.44, 2.41)

7.61 (0.43, 133.33)

17.81 (1.06, 298.38)

ES (95% CI)

9.32 (0.59, 148.16)

1.60 (0.67, 3.80)

8.68 (1.26, 59.95)

3.00 (0.14, 65.90)

1.42 (0.89, 2.26)

5.98 (1.45, 24.71)

2.10 (0.11, 40.70)

1.73 (0.57, 5.22)

2.04 (0.67, 6.21)

7.00 (0.40, 122.44)

3.79 (1.06, 13.60)

1.35 (0.74, 2.47)

3.49 (1.11, 10.95)

1.08 (0.51, 2.32)

11.00 (1.67, 72.40)

100.00

0.80

0.83

Weight

%

0.86

8.77

1.77

0.69

30.37

3.28

0.75

5.40

5.32

0.81

4.04

18.04

5.05

11.36

1.86

1.1 1 10 100

logimor ~ N(0,2^2)

Page 30: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

30

Proposal: 4 sensitivity analysesIMORs Options

(e.g.)Sensitive to: Works via:

fixed equal

imor(2 2) Imbalance in missingness

Point estimates

fixed opposite

imor(2 1/2) Amount of missing data

random equal

sdlogimor(2) corr(1)

Imbalance in missingness

Weightings

random uncorrelated

sdlogimor(2) corr(0)

Amount of missing data

Page 31: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

31

Summary• Tool for sensitivity analysis• Requires thought about plausible missing data

mechanisms• Would be nice to overlay sensitivity analysis

with ACA• Further work includes combining uncertainty

with reasons• I also have a program mvmeta for

multivariate meta-analysis

Page 32: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

32

References• 1st part: Higgins JPT, White IR, Wood A. Imputation methods

for missing outcome data in meta-analysis of clinical trials. Clinical Trials, submitted.

• 2nd part: White IR, Higgins JPT, Wood AM. Allowing for uncertainty due to missing data in meta-analysis. 1. Two-stage methods. Statistics in Medicine, in press.

• Related: White IR, Welton NJ, Wood AM, Ades AE, Higgins JPT. Allowing for uncertainty due to missing data in meta-analysis. 2. Hierarchical models. Statistics in Medicine, in press.

• metamiss.ado available from http://www.mrc-bsu.cam.ac.uk/BSUsite/Software/Stata.shtml

Page 33: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

33

Extra slides

Page 34: 1 Meta-analysis with missing data: metamiss Ian White and Julian Higgins MRC Biostatistics Unit, Cambridge, UK Stata users’ group, London 10 September

34

Overall (I-squared = 16.7%, p = 0.257)

Vichaiya

Howard

Nishikawa_82

Spencer

Reschke

Serafetinides

Selman

Garry

Borison_92

Mander

Bechelli

Simpson

Nishikawa_84

Study ID

Durost

Beasley

Chouinard

Arvantis

2.02 (1.51, 2.70)

19.00 (1.13, 318.79)

2.04 (0.67, 6.21)

3.00 (0.14, 65.90)

11.00 (1.67, 72.40)

3.79 (1.06, 13.60)

8.40 (0.50, 140.57)

1.48 (0.37, 5.90)

1.75 (0.52, 5.92)

7.00 (0.40, 122.44)

1.36 (0.68, 2.72)

6.21 (1.35, 28.50)

2.35 (0.12, 44.54)

9.20 (0.52, 162.90)

ES (95% CI)

8.68 (1.26, 59.95)

1.05 (0.34, 3.24)

3.49 (1.11, 10.95)

1.42 (0.86, 2.33)

100.00

1.05

6.75

0.88

2.36

5.13

1.05

4.39

5.63

1.02

17.36

3.60

0.97

1.01

Weight

2.24

6.58

6.40

33.58

%

2.02 (1.51, 2.70)

19.00 (1.13, 318.79)

2.04 (0.67, 6.21)

3.00 (0.14, 65.90)

11.00 (1.67, 72.40)

3.79 (1.06, 13.60)

8.40 (0.50, 140.57)

1.48 (0.37, 5.90)

1.75 (0.52, 5.92)

7.00 (0.40, 122.44)

1.36 (0.68, 2.72)

6.21 (1.35, 28.50)

2.35 (0.12, 44.54)

9.20 (0.52, 162.90)

ES (95% CI)

8.68 (1.26, 59.95)

1.05 (0.34, 3.24)

3.49 (1.11, 10.95)

1.42 (0.86, 2.33)

100.00

1.05

6.75

0.88

2.36

5.13

1.05

4.39

5.63

1.02

17.36

3.60

0.97

1.01

Weight

2.24

6.58

6.40

33.58

%

1.1 1 10 100

Gamble-Hollis