Journal of the Neurological Sciences 287 S1 (2009) S50–S55
Prognosis of the individual course of disease: the elements of time, hetereogeneity
and precision
Martin Daumer
a,
*, Anneke Neuhaus
a
, Joseph Herbert
b
, George Ebers
c
a
Sylvia Lawry Centre for Multiple Sclerosis Research, Munich, Germany
b
NYU Hospital for Joint Diseases, New York, USA
c
Department of Clinical Neurology, University of Oxford, Oxford, United Kingdom
article info
Keywords:
Multiple sclerosis
Clinical trials
Actimetry
Disease progression
Outcome measure
Prognosis
summary
There is no gold standard in monitoring disease activity for clinical trials in multiple sclerosis.
Various outcome measures, including relapses, disability and magnetic resonance imaging (MRI)
measures have been used to demonstrate the efficacy of the different available therapies for
multiple sclerosis. Recently, the potential limitations of these measures have received increasing
attention, and these have stimulated research into more appropriate and sensitive outcome
measures for clinical trials. For example, it has been shown that widely-used MRI measures add
little, if any, independent information to that provided by more clinically relevant measures such
as relapses and disability. Similarly, the Expanded Disability status Scale (EDSS), which is the most
widely-used measure of disability related to multiple sclerosis, is insufficiently sensitive to detect
robust changes in disability over the timeframes usually used in clinical trials. An alternative to
the EDSS is the Multiple Sclerosis Severity Score (MSSS), a severity scale which relates clinical
disability to disease duration. The MSSS was originally developed from a database of nearly ten
thousand patients from eleven European countries and Australia and has since been reproduced
in an independent dataset of 1134 patients from the placebo arms of randomised clinical trials.
Based on the MSSS score, disease severity can be defined, which shows stability over time and
may provide evidence-based decision support for patient management. Another alternative to
measure disability is the objective quantification of physical activity. There is evidence that recent
developments in pervasive computing using tiny accelerometers may have the potential to increase
the reliability and precision of motor assessment, especially in the mid-range of the EDSS. The
outcome measures discussed have potential use as online tools for evidence-based decision support
which are increasingly being used in medical research and clinical decision-making.
© 2009 Elsevier B.V. All rights reserved.
Introduction
Randomised clinical trials of disease-modifying agents in multiple
sclerosis have yielded important information on the potential
benefits of new therapies and have provided data that is sufficiently
robust to demonstrate the efficacy of six such therapies, which have
been approved for the treatment of multiple sclerosis on this basis.
Nonetheless, for a number of clinically relevant endpoints, and in
particular for disability, data generated by these trials have not been
interpretable in a way that is universally accepted. The reasons for
this are likely to relate to statistical power or to endpoints used in
clinical trials. Indeed, given the heterogeneous presentation of the
disease and its extremely variable clinical course, it is difficult to
detect subtle treatment effects over the relatively short, in terms
*Corresponding author. Martin Daumer. Sylvia Lawry Centre for
MS Research, Hohenlindener Str. 1, 81677 Munich, Germany.
Tel.: +49 89 2060 269 50; fax: +49 89 2060 269 51.
E-mail address: daumer@slcmsr.org (M. Daumer).
of the course of the disease, duration of placebo-controlled trials
(generally two years or less) in populations of generally <200
patients per arm. In addition, the sensitivity of certain evaluation
scales may be inadequate to discriminate pertinent treatment
effects from random noise [1]. For these reasons, it would be useful
to have a patient database that is sufficiently large and well-
calibrated to assess the sensitivity of outcome measures and to
model the evolution over time of critical disease attributes such
as disability. Since 2001, the Sylvia Lawry Centre for Multiple
Sclerosis Research (SLCMSR) in Munich has been assembling such
a database and using it to evaluate the pertinence of different
outcome measures.
The Ian McDonald MS database
This database was established as an initiative of the Multiple
Sclerosis International Federation (MSIF), who wished to fund an
international research and resource centre at which sophisticated
statistical methods could be used to identify clinical and magnetic
0022-510X/ $ – see front matter © 2009 Elsevier B.V. All rights reserved.