Study Designs. Simon Day, PhD Johns Hopkins University



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This work is licensed under a Creative Commons Attribution-NonCommercial-ShareAlike License. Your use of this material constitutes acceptance of that license and the conditions of use of materials on this site. Copyright 2008, The Johns Hopkins University and Simon Day. All rights reserved. Use of these materials permitted only in accordance with license rights granted. Materials provided AS IS ; no representations or warranties provided. User assumes all responsibility for use, and all liability related thereto, and must independently review all materials for accuracy and efficacy. May contain materials owned by others. User is responsible for obtaining permissions for use from third parties as needed.

Study Designs Simon Day, PhD Johns Hopkins University

Section A Treatment Allocation Method; Blinding of Assigned Treatment

Allocation History Earliest trials used arbitrary methods based on convenience Prior case series Patients treated in different sites Arbitrary division of concurrently appearing points More systematic methods in early 20th Century Alternating patients Alphabetical Day of the months Randomization paradigm shift 4

Treatment Allocation Allocation variations: Stratified Blocked Group (cluster) Adaptive 5

Blinding/Masking Blinding limits bias Patient response Physician attitude Outcome evaluation Decision-making Not all trials can be blinded Distinctive side effect(s) Method of administration Grey areas Placebo injections Extra blood draws Sham surgery 6

Blinding/Masking Single blind: patient doesn t know treatment Double blind: patient and treating physician Triple blind: patient, treating physician and outcomes evaluator Unblinded or open label: usually refers to patient and treating physician, can still involve blinded outcome evaluation 7

Blinding/Masking Selection bias Occurs when allocation to one treatment is more likely (predictable) This is why randomization and unpredictability are so important And we need to protect against possible known causes of selection bias and unknown ones 8

Randomization: Why Do It? To Maintain Trial Integrity Produces study groups comparable with respect to known and unknown risk factors Removes possible investigator bias in allocation of participants Guarantees that statistical tests will have valid significance 9

Insoluble Research Problems Lack of representative sampling Imprecise subject selection Non-randomized treatment allocation Inclusion of inappropriate controls Unblinded subjects/evaluators Subjective assessment of outcome 10

Randomization Principles Treatment assignment based on chance alone All subjects are equally likely to be assigned to any group Reduces systematic differences Subjects in each group can be expected to possess essentially the same characteristics 11

Identical Baseline Characteristics The ideal experimental model for comparing two treatments is one in which the baseline characteristics of the two study groups are identical in all aspects 12

Sample Flowchart Population at large Population w/o condition Definition of condition With condition but ineligible Eligible but not enrolled Population with condition Study population Study sample Entry criteria Enrollment And these we randomize 13

Effect of Randomization: Simple Randomization All subjects n=120 30 30 30 30 14

Effect of Randomization: Stratified Randomization All subjects 100 males/20 females 40 young/80 old 20 healthy/100 less healthy 25 males/5 females 10 young/20 old 5 healthy/25 less 25 males/5 fem. 10 young/20 old 5 healthy/25 less 25/5 10/20 5/25 25/5 10/20 5/25 15

Post-Randomization Unknowns Differential group: Compliance Drop-out/lost-to-follow-up Adverse events Trial sabotage/unblinding Other unforeseen issues (different treatment effects!) 16

Suggestions from CONSORT* Assignment/allocation Describe Unit of randomization (person, cluster, etc.) Method used to generate allocation schedule Method of allocation concealment and timing of assignment Method used to separate generator from executor of assignment Source: Begg, C., et. al. (August 28, 1996). Improving the Quality of Reporting RCTs. JAMA. Vol. 276 (8): 637-9 *Note: Consolidation of Standards for Reporting Trials 17

Random Allocation In prospective randomized clinical trials, there is typically a single sample of patients chosen by a non-random set of methods, and THEN random allocation is employed for treatment assignment By calling our methods random allocation We also describe Bias-free treatment assignment 18

Requirements of Good Allocation Schemes Assignment remains masked ( blinded ) to the patient and all study personnel Future assignments can not be predicted The order of allocations is reproducible The generation process has known mathematical properties The process provides a clear audit trail 19

Methods with Problems Coin toss Odd-even schemes (days, dates, time of day) Matched samples Stratification Patient characteristics (date of birth, social security number, medical record number, etc.) Systematic (every nth patient) Sealed envelopes with assignment codes 20

Allocation Management Code generation Local (single site) Local (multiple sites) Central coordination (for multiple sites) Location of masterfile and logs 21

Allocation Management Site location of codes Clinic envelopes Pharmacy On-site computerized generation On-site data center personnel Telephone or computer link to coordinating centre Issues Mechanism for updating enrollment database Maintenance of computer/file security 22

In the Next Section We ll Look at... More on treatment allocation Blocking Stratification Adaptive randomization Unequal sample size 23

Section B Stratification

Definitions Stratified/strata: levels of a risk factor or the categorization of risk factors into different levels Blocks: size of string desired for balancing patient enrollment among trt groups Permuted: randomly assorted Permuted block: randomly assorted blocks of a specific size Image source: www.sxc.hu 25

Allocation Schemes Fixed Simple Blocked Stratified Simple random within strata Permuted block by strata Adaptive Baseline adaptive (aka: minimization) Response/outcome (Zelen: play-the-winner) Number (uses a priori group allocation ratios) 26

Fixed Allocation Fixed allocation assigns the intervention to participants with a prescribed probability (usually equal) Allocation types Simple allocation Blocked allocation Stratified allocation 27

Fixed Allocation Simple Coin toss Random number table Uniform random number generator Problem Imbalance in treatment allocations can occur and vary in severity depending on the overall sample size 28

Simple Allocations Possible imbalance in simple randomization with two treatments. This table shows the difference in treatment numbers (or more extreme) liable to occur with probability at least 0.05 or at least 0.01 for various trial sizes. Difference in numbers Total number of patients Probability 0.05 Probability 0.01 10 2:8 1:9 20 6:14 4:16 50 18:32 16:34 100 40:60 37:63 200 86:114 82:118 500 228:272 221:279 1,000 469:531 459:541 29

Fixed Allocation Blocked A technique which guarantees that at no time during randomization will the imbalance in patient treatment assignment be large, and that at certain times, the number allocated to each group will be equal Translation: choose where to have balance, after every nth enrolled subject (e.g., after every 4th, 6th, 10th) This number of patients is then called the block size or blocking factor 30

Reasons for Blocked Schemes Serial or seasonal variation in severity of illness within likely subject pool Change in standard of care Radiological (e.g., x-ray/ct vs. MRI) Medical Psychological Long-term community enrollments Accumulating outcome information 31

Blocked Randomization Advantage Balance between the number of participants in each study group is guaranteed during the course of the trial Common disadvantage If trial is not blinded, as the end of the block size is reached, the blocking scheme can often be detected and a certain proportion of randomization assignments will be known Solution: use variable block sizes (usually randomly assorted) 32

Fixed Allocations An example of permuted blocks using a block size of four with two treatments, A and B The following represents all of the unique ways that four people could be assigned to two treatments such that half of them get assigned to A and the other half to B (i.e., the permutations of the sequence): AABB ABAB ABBA BBAA BABA BAAB 33

Fixed Allocation Problem AABB ABAB ABBA Problem: if study personnel detect the size of the block (four), they will always know what the last person in a block will receive (e.g., if AAB_ from AABB sequence has already been assigned, it is obvious the next person would receive B) Solution: randomize the size of the subsequent block after each current block is used; from the above, after using the first block of four, the next block size could be randomly selected from sized two, four, or eight 34

Stratification Notes 1. Although simple randomization within strata can be used, blocked randomization is preferred for balance 2. In multi-center trials, the center itself is generally used as one of the stratification variables 3. Controversy remains regarding the need for intra-center stratification on potential risk factors Generally can be handled analytically Design issues and power must be carefully considered All must be pre-specified 35

Adaptive Randomization A technique by which subjects are assigned to a group in a manner that will tend to correct an existing imbalance or cause the least imbalance in prognostic factors Types Baseline Response/outcome These techniques can utilize either deterministic or probabilistic schemes 36

Adaptive Randomization The next patient can be assigned not in a completely random way, but such that whichever group they are assigned to minimizes the overall difference between the groups (i.e., makes them as similar as possible) Not technically difficult Practically can be very difficult! If you do not use a computerized algorithm to make such assignments, for some of these schemes you will get completely lost and reproducibility would be impossible to claim 37

Special Procedure Minimization: special case of general adaptive randomization where allocation procedure is deterministic This means that no chance (randomness) is involved The next patient in a series will simply receive an assignment based on the allocation which reduces any existing imbalance among the groups If no imbalance exists at a given point of assignment, then only that particular patient is randomly allocated 38

Example (Pocock, 1983) Factor Level Number on each treatment A B Next patient Performance status Ambulatory 30 31 Non-ambulatory 10 9 Age <50 18 17 50 22 23 Disease-free interval <2 years 31 32 2 years 9 8 Dominant metastatic Visceral 19 21 Lesion Osseous 8 7 Soft tissue 13 12 39

Example (Pocock, 1983) Factor Level Number on each treatment A B Next patient Performance status Ambulatory 30 31 Non-ambulatory 10 9 Age <50 18 17 50 22 23 Disease-free interval <2 years 31 32 2 years 9 8 Dominant metastatic Visceral 19 21 Lesion Osseous 8 7 Soft tissue 13 12 Totals 76 77 40

Other Randomization Issues Important to consider the timing of randomization relative to the start of treatment; this must always be obvious in protocol Variant: randomized consent scheme (Zelen) patient eligible randomize Do not seek consent Seek consent (accept treatment B?) allocate to treatment A NO YES allocate to allocate to treatment A treatment B Source: Zelen 41

Statistical Efficiency of Treatment Comparisons Reduction in Power of a Trial as the Proportion on the New treatment Is Increased Statistical efficiency of treatment comparisons when a 1:1 randomization scheme is not used Recommendation: if possible, always use 1:1 allocation, but if necessary for assessment of new, standard treatments, use ratio less extreme than 3:1 Power 1 0.9 0.8 0.7 0.6 0.5 0.4 0.3 0.2 0.1 0 50% 60% 70% 80% 90% 100% Percentage of Patients on New Treatment Source: Pocock, S. (1983). Clinical Trials. 42

Questions How do you decide what to do in your study? Should the allocation ration be 1:1 (i.e., 50/50) or some other ratio? What about if you have a manufacturing problem? 43

In the Next Lecture We ll Look at... Control groups Reasons Types of control No treatment-control, placebo control, active control Historical controls Concurrent controls 44