ProBio: Assessing Operating Characteristics via Simulations

Alessio Crippa
Department of Medical Epidemiology and Biostatistics
Karolinska Institutet

Outline


  • Describe the design of the study and illustrate the simulation settings

  • Present the preliminary results (N = 200)

  • Explicit the hypothesis

  • Next steps

Aim

Determine if treatment assignment based on a biomarker signature can improve Progression Free Survival (PFS) compared to standard of care

  • 6 groups:
    • 5 treatments: Enzalutamide, Abiraterone, Radium 223, Cabazitaxel, Docetaxel
    • 1 control which is a mix of the 5 treatments (standard of care)
  • 4 binary biomarkers: Androgen receptor alterations (ARA), DNA-repair deficiency (DRD), TP53 mutations, TMPRSS2-ERG gene fusion (TEfus)
  • The biomarker subtypes are a subset of the (42 = 16) combinations of the 4 biomarkers
   ARA DRD TP53 TEfus prev (%)
1    -   +    +     +      0.5
2    -   -    +     +     15.0
3    +   +    -     +      5.0
4    -   +    -     +      0.5
5    +   -    -     +      7.0
6    -   -    -     +      4.0
7    -   +    +     -      3.0
8    -   -    +     -     18.0
9    +   +    -     -      7.0
10   -   +    -     -      3.0
11   +   -    -     -     32.0
12   -   -    -     -      5.0

Biomarker Signatures

  • A limited number of disease subtypes based on the 4 biomarkers
  • Among 12 possible combinations of the 4 biomarkers, we are interested in 9 signatures
  signatures -+++ --++ ++-+ -+-+ +--+ ---+ -++- --+- ++-- -+-- +--- ---- prev
1        all    X    X    X    X    X    X    X    X    X    X    X    X 1.00
2       ARA+              X         X                   X         X      0.51
3       ARA-    X    X         X         X    X    X         X         X 0.49
4       DRD+    X         X    X              X         X    X           0.19
5       DRD-         X              X    X         X              X    X 0.81
6      TP53+    X    X                        X    X                     0.36
7      TP53-              X    X    X    X              X    X    X    X 0.64
8     TEfus+    X    X    X    X    X    X                               0.32
9     TEfus-                                  X    X    X    X    X    X 0.68

Simulation Algorithm

Enter participants in a simulated trial:

  1. Simulate partipants' subtypes (based on observed prevalences)

  2. Assign treatment within each subtype, according to probability of superiority (initially equal randomization)
    The control group had probabilities 33%, 33%, 11%, 11%, 11% of receiving treatment Enzalutamide, Abiraterone, Radium 223, Cabazitaxel, Docetaxel respectively

  3. Simulate outcome: PFS according to selected scenarios

  4. Update probability of superiority within subtypes

  5. Update probability of superiority within signatures (deciding for early stopping)

  6. Update randomization probabilities based on the quantities calculated in 4


Repeat 1-6 until stopping rules are met, or after reaching maximum number of patients (100) within each signature

Maximum trial duration (24 months)

Parametric models for survival analysis

\( T_{ixk} \sim Exp(\lambda_{xk}) \)
\( i = 1, \dots, n_{xk}, \;\; \) \( x = 2, \dots, 6 \; \) \( k = 1, \dots, 12 \)

  • Distributional assumption: \( \mu_{xk} = \frac{1}{\lambda_{xk}} \sim IG(\alpha_{xk}, \beta_{xk}) \)

  • Observed data: censored survival times \( t_{ixk} \) and event indicators \( \delta_{ixk} \)

  • Update parameters of prior distribution: \( \alpha_{xk}' = \alpha_{xk} + \sum_i \delta_{ixk} \) and \( \beta_{xk}' = \beta_{xk} + \sum_i t_{ixk} \)

  • Compare the mean PFS times \( \mu_{xk} \) within each subtype by computing (update randomization probabilities)
    \( p_{xk} = Pr(\mu_{xk} - \mu_{1k} \ge d | data) \)
    \( x = 2, \dots, 6 \), \( k = 1, \dots, 12 \)

  • Compare the mean PFS times \( \mu_{xl} \) within each signature by computing (deciding for early stopping) \( p_{xl} = Pr(\mu_{xl} - \mu_{1l} \ge d | data) \)
    \( x = 2, \dots, 6 \), \( l = 1, \dots, 9 \)

Stopping rules

  1. Futility:

    • \( n_{xl} \ge 20 \)
    • \( p_{xl} \le 0.1 \; \forall l = 1, \dots, 9 \)
  2. Graduation:

    • \( n_{xl} \ge 50 \)
    • \( p_{xl} \ge 0.85 \)
    • \( p_{xk} \ge 0.25 \; \forall l = 1, \dots, 9 \) and \forall k \in l$
  3. Stop randomization:

    • \( n_{xl} \ge 100 \)
    • month \( \ge 24 \)

Example graduation: Radium 223 for ARA+ patients

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all           0.002       0.000      0.164       0.090     0.000
ARA+          0.007       0.082      0.981       0.299     0.005
ARA-          0.003       0.000      0.003       0.092     0.008
DRD+          0.022       0.128      0.244       0.413     0.059
DRD-          0.001       0.000      0.229       0.043     0.004
TP53+         0.012       0.010      0.008       0.195     0.011
TP53-         0.004       0.007      0.623       0.144     0.007
TEfus+        0.046       0.045      0.210       0.475     0.019
TEfus-        0.000       0.000      0.269       0.029     0.004
     Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
-+++        0.299       0.419      0.321       0.408     0.346
--++        0.076       0.003      0.007       0.166     0.000
++-+        0.276       0.789      0.404       0.942     0.447
-+-+        0.165       0.106      0.175       0.135     0.165
+--+        0.603       0.591      0.959       0.727     0.288
---+        0.091       0.150      0.388       0.711     0.195
-++-        0.280       0.274      0.323       0.691     0.213
--+-        0.009       0.033      0.002       0.049     0.186
++--        0.017       0.299      0.824       0.172     0.036
-+--        0.227       0.152      0.156       0.376     0.382
+---        0.000       0.000      0.929       0.008     0.000
----        0.204       0.089      0.123       0.146     0.195

Simulation scenarios


  1. No treatment effective in any subtype

  2. Radium 223 effective in all subtypes, other treatments ineffective

  3. Radium 223 effective for ARA+ (prev = 0.51), other treatments ineffective

  4. Enzalutamide effective for ARA+, other treatments ineffective

  5. As in 4., but with Enzalutamide representing 75% of the control group

Table results


  1. Assumed PFS times (and definition of control group)

  2. Average number of enrolled participants

  3. Graduation times for each treatments
    Power (percentage of times we graduated a treatment for the true biomarker signature)
    False-positive rate (percentage of times that a treatment graduated only for a signature that didn't benefit)

  4. Graduation times for each signature

  5. Probability of superiority for each signature

Results 1: No treatment effective in any subtype


True PFS times

$mi_gx
     Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
-+++            6           6          6           6         6
--++            6           6          6           6         6
++-+            6           6          6           6         6
-+-+            6           6          6           6         6
+--+            6           6          6           6         6
---+            6           6          6           6         6
-++-            6           6          6           6         6
--+-            6           6          6           6         6
++--            6           6          6           6         6
-+--            6           6          6           6         6
+---            6           6          6           6         6
----            6           6          6           6         6

$p_ctr
Enzalutamide  Abiraterone   Radium 223  Cabazitaxel    Docetaxel 
        0.33         0.33         0.11         0.11         0.11 

Average Number of Participants by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all             161         164        162         164       164
ARA+             80          79         79          81        78
ARA-             81          84         83          82        86
DRD+             80          79         79          81        78
DRD-             81          84         83          82        86
TP53+            61          64         63          63        65
TP53-           101          99         98         100        99
TEfus+           57          59         57          59        59
TEfus-          104         104        105         104       105

Graduation Probabilities

             p1    p2
Enzalutamide  0 0.075
Abiraterone   0 0.075
Radium.223    0 0.040
Cabazitaxel   0 0.040
Docetaxel     0 0.055

Graduation Times by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all              24          24         24          24        24
ARA+             23          23         22          23        23
ARA-             24          24         24          24        24
DRD+             24          24         24          24        24
DRD-             24          24         24          24        24
TP53+            24          24         24          24        24
TP53-            24          24         24          24        24
TEfus+           24          24         24          24        24
TEfus-           24          24         24          24        24

Probability of superiority by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all            0.08       0.072      0.063       0.072     0.083
ARA+           0.19       0.182      0.160       0.156     0.182
ARA-           0.12       0.113      0.106       0.122     0.126
DRD+           0.15       0.144      0.139       0.146     0.172
DRD-           0.15       0.150      0.111       0.137     0.134
TP53+          0.17       0.169      0.179       0.200     0.194
TP53-          0.13       0.119      0.095       0.104     0.118
TEfus+         0.15       0.149      0.142       0.154     0.166
TEfus-         0.14       0.130      0.114       0.129     0.138

Results 2: Radium 223 effective in all subtypes, other treatments ineffective


True PFS times

$mi_gx
     Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
-+++            6           6         12           6         6
--++            6           6         12           6         6
++-+            6           6         12           6         6
-+-+            6           6         12           6         6
+--+            6           6         12           6         6
---+            6           6         12           6         6
-++-            6           6         12           6         6
--+-            6           6         12           6         6
++--            6           6         12           6         6
-+--            6           6         12           6         6
+---            6           6         12           6         6
----            6           6         12           6         6

$p_ctr
Enzalutamide  Abiraterone   Radium 223  Cabazitaxel    Docetaxel 
        0.33         0.33         0.11         0.11         0.11 

Average Number of Participants by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all             161         163        143         162       161
ARA+             75          76         72          77        75
ARA-             86          86         71          85        87
DRD+             75          76         72          77        75
DRD-             86          86         71          85        87
TP53+            64          65         57          64        65
TP53-            97          98         86          98        96
TEfus+           58          59         51          60        60
TEfus-          103         103         92         102       102

Graduation Probabilities

               p1    p2
Enzalutamide 0.00 0.040
Abiraterone  0.00 0.025
Radium.223   0.88 0.000
Cabazitaxel  0.00 0.055
Docetaxel    0.00 0.040

Graduation Times by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all              24          24         22          24        24
ARA+             22          22         17          23        22
ARA-             24          24         21          24        24
DRD+             24          24         23          24        24
DRD-             24          24         17          24        23
TP53+            24          24         20          24        24
TP53-            24          24         22          24        24
TEfus+           24          24         22          24        24
TEfus-           24          24         19          24        23

Probability of superiority by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all           0.017       0.011       0.97       0.017    0.0084
ARA+          0.051       0.063       0.95       0.066    0.0500
ARA-          0.048       0.033       0.87       0.048    0.0330
DRD+          0.086       0.083       0.63       0.093    0.0919
DRD-          0.030       0.018       0.99       0.034    0.0186
TP53+         0.083       0.069       0.89       0.098    0.0638
TP53-         0.034       0.032       0.91       0.032    0.0294
TEfus+        0.063       0.056       0.84       0.081    0.0589
TEfus-        0.043       0.047       0.94       0.043    0.0318

Results 3: Radium 223 effective for ARA+ (prev = 0.51), other treatments ineffective


True PFS times

$mi_gx
     Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
-+++            6           6          6           6         6
--++            6           6          6           6         6
++-+            6           6         12           6         6
-+-+            6           6          6           6         6
+--+            6           6         12           6         6
---+            6           6          6           6         6
-++-            6           6          6           6         6
--+-            6           6          6           6         6
++--            6           6         12           6         6
-+--            6           6          6           6         6
+---            6           6         12           6         6
----            6           6          6           6         6

$p_ctr
Enzalutamide  Abiraterone   Radium 223  Cabazitaxel    Docetaxel 
        0.33         0.33         0.11         0.11         0.11 

Average Number of Participants by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all             163         162        153         162       163
ARA+             76          76         78          77        77
ARA-             88          86         75          86        86
DRD+             76          76         78          77        77
DRD-             88          86         75          86        86
TP53+            67          64         58          64        65
TP53-            97          98         94          98        98
TEfus+           61          58         52          59        61
TEfus-          102         104        101         103       102

Graduation Probabilities

              p1    p2
Enzalutamide 0.0 0.040
Abiraterone  0.0 0.040
Radium.223   0.6 0.030
Cabazitaxel  0.0 0.035
Docetaxel    0.0 0.050

Graduation Times by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all              24          24         24          24        24
ARA+             22          23         15          23        23
ARA-             24          24         24          24        24
DRD+             24          24         23          24        24
DRD-             24          24         23          24        24
TP53+            24          24         24          24        24
TP53-            24          24         23          24        24
TEfus+           24          24         24          24        24
TEfus-           24          24         23          24        24

Probability of superiority by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all           0.032       0.038       0.58       0.042     0.026
ARA+          0.069       0.056       0.95       0.071     0.054
ARA-          0.108       0.117       0.12       0.127     0.104
DRD+          0.117       0.113       0.40       0.147     0.105
DRD-          0.071       0.072       0.68       0.072     0.058
TP53+         0.181       0.178       0.20       0.199     0.168
TP53-         0.047       0.050       0.77       0.056     0.044
TEfus+        0.116       0.121       0.40       0.141     0.092
TEfus-        0.070       0.068       0.69       0.076     0.074

Results 4: Enzalutamide effective for ARA+ (prev = 0.51), other treatments ineffective


True PFS times

$mi_gx
     Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
-+++            6           6          6           6         6
--++            6           6          6           6         6
++-+           12           6          6           6         6
-+-+            6           6          6           6         6
+--+           12           6          6           6         6
---+            6           6          6           6         6
-++-            6           6          6           6         6
--+-            6           6          6           6         6
++--           12           6          6           6         6
-+--            6           6          6           6         6
+---           12           6          6           6         6
----            6           6          6           6         6

$p_ctr
Enzalutamide  Abiraterone   Radium 223  Cabazitaxel    Docetaxel 
        0.33         0.33         0.11         0.11         0.11 

Average Number of Participants by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all             160         156        154         154       156
ARA+             83          73         73          72        73
ARA-             78          83         81          82        82
DRD+             83          73         73          72        73
DRD-             78          83         81          82        82
TP53+            62          62         60          62        61
TP53-            98          95         94          92        95
TEfus+           57          56         56          53        55
TEfus-          103         101         99         101       100

Graduation Probabilities

               p1    p2
Enzalutamide 0.41 0.055
Abiraterone  0.00 0.005
Radium.223   0.00 0.010
Cabazitaxel  0.00 0.030
Docetaxel    0.00 0.015

Graduation Times by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all              24          24         24          24        24
ARA+             18          21         21          21        21
ARA-             24          24         24          24        24
DRD+             24          24         24          24        24
DRD-             24          22         22          22        22
TP53+            24          24         24          23        24
TP53-            23          24         24          24        24
TEfus+           24          24         24          24        24
TEfus-           23          24         23          24        23

Probability of superiority by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all            0.65       0.034      0.032       0.032     0.033
ARA+           0.97       0.049      0.065       0.054     0.055
ARA-           0.13       0.121      0.100       0.117     0.120
DRD+           0.41       0.130      0.123       0.115     0.124
DRD-           0.78       0.069      0.066       0.076     0.066
TP53+          0.22       0.173      0.163       0.190     0.168
TP53-          0.81       0.045      0.049       0.042     0.043
TEfus+         0.51       0.111      0.104       0.108     0.099
TEfus-         0.69       0.072      0.071       0.075     0.077

Results 5: As in 4., but with Enzalutamide representing 75% of the control group


True PFS times

$mi_gx
     Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
-+++            6           6          6           6         6
--++            6           6          6           6         6
++-+           12           6          6           6         6
-+-+            6           6          6           6         6
+--+           12           6          6           6         6
---+            6           6          6           6         6
-++-            6           6          6           6         6
--+-            6           6          6           6         6
++--           12           6          6           6         6
-+--            6           6          6           6         6
+---           12           6          6           6         6
----            6           6          6           6         6

$p_ctr
Enzalutamide  Abiraterone   Radium 223  Cabazitaxel    Docetaxel 
       0.750        0.083        0.056        0.056        0.056 

Average Number of Participants by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all             158         146        142         147       144
ARA+             89          58         55          60        58
ARA-             69          88         87          87        86
DRD+             89          58         55          60        58
DRD-             69          88         87          87        86
TP53+            56          63         63          64        64
TP53-           102          83         79          83        80
TEfus+           56          54         54          56        54
TEfus-          102          91         88          90        90

Graduation Probabilities

               p1    p2
Enzalutamide 0.23 0.005
Abiraterone  0.00 0.000
Radium.223   0.00 0.000
Cabazitaxel  0.00 0.000
Docetaxel    0.00 0.000

Graduation Times by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all              24          24         24          24        24
ARA+             20          17         16          16        16
ARA-             24          24         24          24        24
DRD+             24          24         24          24        24
DRD-             24          23         22          23        23
TP53+            24          24         24          24        24
TP53-            24          24         24          24        24
TEfus+           24          24         24          24        24
TEfus-           24          23         22          22        22

Probability of superiority by Signature

       Enzalutamide Abiraterone Radium 223 Cabazitaxel Docetaxel
all            0.65       0.037      0.033       0.038     0.028
ARA+           0.97       0.052      0.038       0.058     0.042
ARA-           0.12       0.131      0.128       0.137     0.119
DRD+           0.45       0.125      0.109       0.109     0.101
DRD-           0.73       0.067      0.071       0.089     0.066
TP53+          0.22       0.182      0.181       0.195     0.176
TP53-          0.81       0.048      0.038       0.046     0.036
TEfus+         0.53       0.099      0.109       0.120     0.097
TEfus-         0.67       0.091      0.065       0.075     0.074

Simulation parameters and setting

  1. PFS scenarios:

    • effect size 12 vs 6 months
  2. General settings:

    • accrual rate of 60 patients/month
    • biomarker subtypes prevalences
    • length of follow-up (2 years)
    • priors (\( \alpha = 11 \), \( \beta = 60 \))
  3. Decision rules:

    • probabilities: pU = 0.85, pU2 = 0.25, pL = 0.1
    • number of patients: nmin = 20, nval = 50, nmax = 100
    • minimum difference: d = 1 month
  4. Number of simulations: N = 200

Next steps

Extend simulation to investigate

  1. SMART design:

    • define parameters for optimal treatments sequences
    • implement adaptive randomization (Q-learning?)
  2. Effectiveness (current practice) vs efficacy:

    • how to compare treatments efficacies