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Biomarkers of Joint Destruction and Repair Objectives ·
Identify
new and promising biomarkers for diseases characterized by progressive
joint destruction ·
Define
the knowledge gaps and research opportunities to advance the understanding
and use of biomarkers as surrogate endpoints in clinical trials for
diseases characterized by progressive joint destruction Agenda
Moderator:
Kenneth D. Brandt, M.D., Indiana University School of Medicine Introductory
Remarks:
Suzanne Serrate-Sztein, M.D., National Institute of Arthritis and
Musculoskeletal and Skin Diseases Session
I. Imaging Technologies Imaging Markers of Joint Destruction and Repair Charles
G. Peterfy, M.D., Ph.D., University of California, San Francisco, and
Synarc, Inc. Imaging Biomarkers in Back Pain Robert
D. Boutin, M.D., University of California and Veterans Affairs Medical
Center, San Diego Imaging Biomarkers in Osteoarthritis Harry
Genant, M.D., University of California, San Francisco Imaging Technology for Osteoarthritis Mark
E. Brezinski, M.D., Ph.D., Massachusetts General Hospital Discussion Session
II. Inflammatory and Genetic
Markers Cytokine Expression in Arthritis Gary
S. Firestein, M.D., University of California, San Diego Genetic and Major Histocompatibility Complex Markers of Disease Severity
in Rheumatoid Arthritis Cornelia
M. Weyand, M.D., Ph.D., Mayo Foundation Discussion Session
III. Biochemical Markers of
Cartilage Breakdown and Repair Serum Markers of Joint Metabolism Eugene
J-M.A. Thonar, Ph.D., Rush Medical College Molecular Markers in Osteoarthritis Stefan
L. Lohmander, M.D., Ph.D., University of Lund, Sweden Biomarkers of Joint Destruction and Repair (continued) Collagen Type II Cross-Linked Telopeptides: A Promising Marker of Cartilage Degradation
in Arthritis David
Eyre, Ph.D., University of Washington Synovial Fluid Markers of Osteoarthritis Kenneth
D. Brandt, M.D. Discussion Summary
and Conclusions
Imaging
Markers of Joint Destruction and Repair Charles
G. Peterfy, M.D., Ph.D. and Harry K. Genant, M.D. The
past two decades have seen remarkable advances in medical imaging.
The development of magnetic resonance imaging, in particular, has
brought unprecedented power to the study of joint disease and its causes
and has offered a unique opportunity to explore arthritis in ways not
imaginable in the past. This
discussion will review the different roles that medical imaging can play
in clinical trials and outline the deliverables that imaging markers must
strive to meet to be useful in these roles.
It also will review the most promising imaging markers available
today for evaluating arthritic changes in bone, articular cartilage, and
other joint structures and point to areas where further progress can be
anticipated. Key
References Altman
RD, Hochberg M, Murphy WAJ, Wolfe F, Lequesne M. Atlas of individual
radiographic features in osteoarthritis. Osteoarthritis Cartilage
1996;3(suppl. A):3‑70. Bashir
A, Gray ML, Burstein D. Gd‑DTPA as a measure of cartilage
degradation. Magn Reson Med 1996;36:665‑673. Buckland‑Wright
JC, Macfarlane DG, Lynch JA, Jasani MK, Bradshaw CR. Joint space width
measures cartilage thickness in osteoarthritis of the knee: High
resolution plain film and double contrast macroradiographic investigation.
Ann Rheum Dis 1995;54:263‑268. Disler
DG, McCauley TR, Kelman CG, Fuchs MD, Ratner LM, Wirth CR, Hospodar PP.
Fat‑suppressed three‑dimensional spoiled gradient‑echo
MR imaging of hyaline cartilage defects in the knee: Comparison with
standard MR imaging and arthroscopy. Am J Rheumatol
1996;167:127‑132. Peterfy
C. Imaging techniques. In: Rheumatology 2d ed. Klippel J, Dieppe P (eds.)
Philadelphia: Mosby 1997;1:14.1-14.18. Peterfy
C. Magnetic resonance imaging. Osteoarthritis. In: Brandt K, Doherty M,
Lohmander L (eds.) New York: Oxford University Press 1998;473-494. Peterfy
CG, Majumdar S, Lang P, van Dijke CF, Sack K, Genant H. MR imaging of the
arthritic knee: Improved discrimination of cartilage, synovium and
effusion with pulsed saturation transfer and fat‑suppressed
T1‑weighted sequences. Radiology 1994;191:413‑419. Recht
MP, Pirraino DW, Paletta GA, Schils JP, Belhobek GH. Accuracy of
fat‑suppressed three‑dimensional spoiled gradient‑echo
FLASH MR imaging in the detection of patellofemoral articular cartilage
abnormalities. Radiology 1996;198:209‑212. Imaging
of Ankylosing Spondylitis Robert
D. Boutin, M.D. Radiological
evaluation of arthropathies is classically based on analysis of two
fundamental findings: the
distribution and morphology of osteoarticular abnormalities.
Ankylosing spondylitis (AS) primarily targets synovial and
cartilaginous joints, as well as sites of tendinous and ligamentous
attachment to bone. Radiographic
abnormalities in patients with AS predominate in the sacroiliac joints and
spine, following in descending order of frequency by the hips,
glenohumeral joints, knees, hands, wrists, and feet.
In the past, scientific investigations commonly evaluated the
efficacy of imaging in providing an accurate diagnosis of early AS.
Although the early and accurate diagnosis of AS is still a primary
goal of imaging, other concerns and questions are voiced with increasing
frequency. For example, given
the substantial constraints imposed by managed care, how should imaging be
utilized to provide a cost‑effective impact on patient care and
outcome? Specifically, what
is the optimal imaging algorithm for the assessment of patients?
Other questions focus on more academic (and corporate) concerns:
What is the most sensitive and specific means for detecting
response to a new therapy? Although
conventional radiography continues to be the principal method of
radiologic assessment, other imaging methods (e.g., magnetic resonance
imaging) are expected to be used in the future with increasing frequency
to detect pathologic changes involving joints, bones, and entheses. Imaging
Technology for Osteoarthritis Mark
E. Brezinski, M.D., Ph.D. A
variety of modalities have now shown feasibility for modifying the
progression of articular cartilage damage in osteoarthritis. This has resulted in a need for better methods to image early
changes in cartilage and monitor the progression of these changes.
The advantages and limitations of traditional imaging methods, such
as radiography, low‑frequency ultrasound, computed tomography,
magnetic resonance imaging, and arthroscopy, for assessing articular
cartilage, will be examined. In
addition, newer technologies, such as optical coherence tomography (OCT),
will be discussed. OCT is a
new method of high‑resolution imaging that allows distances to be
measured on a micron scale. OCT
can be envisioned as analogous to ultrasound, measuring the intensity of
backreflected infrared light rather than sound.
All technologies will be discussed with respect to their ability to
identify fine changes in the cartilage necessary for monitoring
therapeutic modalities. Cytokine
Expression in Arthritis Gary
S. Firestein, M.D. Cytokines
play a key role in the perpetuation of rheumatoid arthritis (RA).
Careful studies of the cytokine profile in RA have demonstrated an
abundance of macrophage and fibroblasts such as IL‑1, TNF‑alpha,
and IL‑6. Furthermore,
production of proinflammatory mediators, including prostaglandins and
metalloproteinases, is regulated in rheumatoid synovium by these
cytokines. In an effort to understand the mechanism of action of
antirheumatic drugs, surrogate markers for clinical trials have been
evaluated. Although
peripheral blood and synovial effusions are more accessible and a few
studies have demonstrated clinical correlations with cytokines or soluble
cytokine receptors, they are probably less useful than direct examination
of synovial tissue. Serial
synovial biopsies to evaluate surrogate markers was first used to examine
the effect of steroids and methotrexate on metalloproteinase gene
expression. Subsequent studies have demonstrated that blind percutaneous
biopsies are essentially equivalent to arthroscopic samples and can be
used to evaluate the expression of cytokines.
For instance, methotrexate and anti‑TNF‑alpha therapy
significantly decrease synovial expression of cytokines as determined by
immunohistochemistry. This method for assessing cytokine expression is reproducible
and has been carefully validated. Attempts
to quantify cytokine mRNA in synovial tissue using nested reverse
transcriptase‑polymerase chain reaction are also promising, although
no studies to date have directly compared mRNA and protein analyses.
Additional studies are required to develop clear criteria for determining
the most appropriate techniques for assaying cytokines as well as the best
cytokine surrogate endpoints. Key
References Dolhain
RJ, Tak PP, Dijkmans BA, De Kuiper P, Breedveld FC, Miltenburg AM.
Methotrexate reduces inflammatory cell numbers, expression of monokines
and of adhesion molecules in synovial tissue of patients with rheumatoid
arthritis. Br J Rheumatol 1998;37:502‑508. Firestein,
GS, Paine M, Boyle DL. Mechanisms of methotrexate action in rheumatoid
arthritis: Selective decrease
in synovial collagenase gene expression. Arthritis Rheum
1994;37:193‑200. Firestein,
GS, Paine M, Littman BH. Gene
expression (collagenase, tissue inhibitor of metalloproteinase,
complement, and HLA‑DR) in rheumatoid arthritis and osteoarthritis
synovium: Quantitative analysis and effect of intra‑articular
corticosteroids. Arthritis Rheum 1991;34:1094‑1105. Kirkham
BW, Navarro FJ, Corkill MM, Panayi GS. In vivo analysis of disease
modifying drug therapy activity in rheumatoid arthritis by sequential
immunohistological analysis of synovial membrane interleukin 1 beta. J
Rheumatol 1994;21:1615‑1619. Kotake
S, Schumacher HR Jr, Yarboro CH, Arayssi TK, Pando JA, Kanik KS, Gourley
MF, Klippel JH, Wilder RL. In vivo gene expression of type 1 and type 2
cytokines in synovial tissues from patients in early stages of rheumatoid,
reactive, and undifferentiated arthritis. Proc Assoc Am Physicians
1997;109:286‑301. Tak
PP, Taylor PC, Breedveld FC, Smeets TJ, Daha MR, Kluin PM, Meinders AE,
Maini RN. Decrease in cellularity and expression of adhesion molecules by
anti‑tumor necrosis factor alpha monoclonal antibody treatment in
patients with rheumatoid arthritis. Arthritis Rheum
1996;39:1077‑1081. Youssef
PP, Kraan M, Breedveld F, Bresnihan B, Cassidy N, Cunnane G, Emery P,
Fitzgerald O, Kane D, Lindblad S, Reece R, Veale D, Tak PP Quantitative
microscopic analysis of inflammation in rheumatoid arthritis synovial
membrane samples selected at arthroscopy compared with samples obtained
blindly by needle biopsy. Arthritis Rheum 1998;41:663‑669. Genetic
and Major Histocompatibility Complex Markers of Disease Severity in
Rheumatoid Arthritis Cornelia
M. Weyand, M.D. Rheumatoid
arthritis (RA) is a chronic inflammatory disease that often leads to
disability and crippling. It
is now recognized that RA is a multigene disorder with several genetic
risk factors contributing to pathogenesis.
We have proposed that multiple subtypes of RA exist and that
different phenotypes of RA correspond to the inheritance of different
arrays of disease risk genes. This
concept has been supported by a detailed analysis of the distribution of
human leukocyte antigen (HLA)‑DRB1 polymorphisms in patient cohorts,
indicating that the clinical heterogeneity in course and outcome
correlates with HLA‑DRB1 allelic polymorphisms.
A set of HLA‑DRB1 alleles has been recognized as disease
associated, but different alleles are enriched in distinct subtypes of RA.
Rheumatoid factor-positive destructive disease is preferentially
associated with the HLA‑DRB1*0401 allele, whereas HLA‑DRB1*0404
and B1*0101 predispose to milder and seronegative disease.
Inheritance of two copies of RA‑associated alleles carries a
high risk for extra‑articular spreading of RA.
Homozygosity for HLA‑DRB1*0401 has been reported in patients
with the most serious complication of RA, rheumatoid vasculitis.
In addition to HLA‑encoded polymorphisms, abnormalities in
the generation and function of CD4 T cells can be useful in dissecting
patient subsets with different variants of RA.
In patients with extra‑articular RA, unusual CD4+ lymphocytes
emerge that are characterized by a deficiency for the CD28 molecule.
CD4+CD28‑ T cells have a tendency to form large clonal
populations, produce high amounts of interferon-gamma, and exhibit
autoreactivity. Accumulation
of these CD4 T cells in a subset of RA patients likely reflects a
fundamental abnormality in T cell homeostasis.
Appreciation of the heterogeneity of the synovial component of RA
has come from studies describing at least three different patterns of
lymphoid organization and tissue cytokine production in the synovium of RA
patients. Genetic elements
determining disease expression in the inflammatory lesions await
identification, but candidate genes include cytokine genes and tissue
injury response genes. The
ultimate goal of these studies is to dissect the phenotypic and genotypic
heterogeneity of RA and to correlate combinations of disease-risk genes
with clinical variants of the disease.
The recognition of clinical subcategories will be required to
optimize pathogenic studies and also provide important tools in the
diagnostic and therapeutic management of patients with the RA syndrome. Key
References Klimiuk
P, Goronzy JJ, Bjornsson J, Beckenbaugh RD, Weyand CM. Tissue cytokine
patterns distinguish variants of rheumatoid synovitis. Am J Pathol
1997;151:1311‑1319. Schmidt
D, Goronzy JJ, Weyand CM. CD4+ CD7‑ CD28‑ T cells are expanded
in rheumatoid arthritis and are characterized by autoreactivity. J Clin
Invest 1996;97:2027‑2037. Wagner
UG, Koetz K, Weyand CM, Goronzy JJ. Perturbation of the T cell repertoire
in rheumatoid arthritis. Proc Natl Acad Sci U S A
1998;95:14447‑14452. Walser‑Kuntz
DR, Weyand CM, Weaver AJ, O'Fallon WM, Goronzy JJ. Mechanisms underlying
the formation of the T cell receptor repertoire in rheumatoid arthritis.
Immunity 1995;2:597‑605. Weyand
CM, Goronzy JJ. The molecular basis of rheumatoid arthritis. J Mol Med
1997;75:772‑785. Weyand
CM, Goronzy JJ. Pathogenesis of rheumatoid arthritis. Med Clin North Amer
1997;81:29‑55. Weyand
CM, Hicok KC, Conn DL, Goronzy JJ. The influence of HLA‑DRB1 genes
on disease severity in rheumatoid arthritis. Ann Intern Med
1992;117:801‑806. Weyand
CM, Klimiuk PA, Goronzy JJ. Heterogeneity of rheumatoid arthritis: From
phenotypes to genotypes. Springer Semin Immunopathol 1998;20:5‑22. Weyand
CM, McCarthy TG, Goronzy JJ. Correlation between disease phenotype and
genetic heterogeneity in rheumatoid arthritis. J Clin Invest
1995;95:2120‑2126. Weyand
CM, Schmidt D, Wagner U, Goronzy JJ. The influence of sex on the phenotype
of rheumatoid arthritis. Arthritis Rheum 1998;41:817‑822. Serum
Markers of Joint Metabolism Eugene
J.‑M. A. Thonar, Ph.D. Fragments
produced when molecules are degraded in joints eventually reach the blood
circulation where they can be measured by sensitive immunoassays.
The levels of these molecular markers provide critical information
concerning the state of health of joint tissues, especially articular
cartilage. Measurements of
the markers in joint disease have already proved useful to monitor changes
in the metabolism of joint tissues following joint injury and to identify
individuals most likely to exhibit rapid joint destruction.
They also can be used to monitor disease progression and to
identify drugs capable of inhibiting deleterious processes or promoting
repair in articular cartilage. Molecular Markers in Osteoarthritis Stefan L. Lohmander, M.D., Ph.D. The
identification of individuals at risk for disease progression, the
monitoring of outcomes in clinical trials, and the treatment of
osteoarthritis (OA) present a challenge.
The lack of readily usable methods hinders progress in the
treatment of joint diseases. Interventions
developed in this area concentrate on agents that inhibit proteolytic
degradation of the cartilage matrix.
Considerable literature exists on molecular markers in human
arthritis and OA (Lohmander et al. 1995, 1998), supporting a relationship
between marker concentrations in joint fluid, serum or urine, and
cartilage turnover. This
provides face validity for these markers to monitor dynamic changes in the
target tissue. Other aspects
of validity for the use of markers are less well supported, but new data
suggest that markers will be useful in future trials.
Increased serum concentrations of hyaluronan, C‑reactive
protein, and cartilage oligomeric matrix protein predict future OA
progression (Månsson et al. 1995; Sharif et al. 1995a, 1995b; Spector et
al. 1997), which can be used to select high‑risk individuals in
early trials. Data on within‑ and between‑patient variability
for molecular markers in joint fluid and serum and urine are available for
stable OA cohorts (Lohmander et al. 1998) and suggest that (1) variability
differs between markers in the same compartment, (2) variability is lower
within than between patients, and (3) markers are responsive to change. Some 30 patients per treatment arm would be needed to show a
change of 0.5 standard deviation with 80‑percent power (Lohmander et
al. 1998). The final answer
on the utility of these surrogate measures must await the availability of
an agent that changes OA disease progression. Key References Lohmander
LS, Dahlberg L, Eyre D, Lark M, Thonar EMJ‑A, Ryd L. Longitudinal
and cross‑sectional variability in markers of joint metabolism in
patients with knee pain and articular cartilage abnormalities.
Osteoarthritis Cartilage 1998;6:351‑361. Lohmander
LS, Felson DT. Defining and validating the clinical role of molecular
markers in osteoarthritis. In: Osteoarthritis. Brandt KD, Doherty M,
Lohmander LS (eds.) Oxford: Oxford University Press, 1998;519-530. Lohmander LS, Saxne T, Heinegård D, editors.
Molecular markers of joint and skeletal diseases. Acta Orthop Scand
1995;66 (suppl):1‑212. Månsson
B, Carey D, Alini M, Ionescu M, Rosenberg LC, Poole AR, Heinegård D,
Saxne T. Cartilage and bone metabolism in rheumatoid arthritis.
Differences between rapid and slow progression of disease identified by
serum markers of cartilage metabolism. J Clin Invest
1995;95:1071‑1077. Sharif
M, George E, Shepstone L, Knudson W, Thonar EJ, Cushnaghan J, Dieppe P.
Serum hyaluronic acid level as a predictor of disease progression in
osteoarthritis of the knee. Arthritis Rheum 1995a;38:760‑767. Sharif
M, Saxne T, Shepstone L, Kirwan JR, Elson CJ, Heinegård D, Dieppe PA.
Relationship between serum cartilage oligomeric matrix protein levels and
disease progression in osteoarthritis of the knee joint. Br J Rheumatol
1995b;34:306‑310. Spector
TD, Hart DJ, Nandra D, Doyle DV, Mackillop N, Gallimore JR, Pepys MB.
Low‑level increases in serum C‑reactive protein are present in
early osteoarthritis of the knee and predict progressive disease.
Arthritis Rheum 1997;40:723‑727. Collagen Type II Cross‑Linked Telopeptides:
A Promising Marker of Cartilage Degradation in Arthritis David Eyre, Ph.D. A
specific cartilage degradation marker has potential value in chondro‑protective
drug development and clinical management of arthritis patients.
There is a need for minimally invasive biochemical assays that can
assess the rate of cartilage degradation in patients with degenerative
joint diseases. The collagen
framework of cartilage turns over extremely slowly in the normal adult,
and its gross degradation in articular cartilage is believed to be a
critical, irreversible event in osteoarthritis.
A degradation assay for cartilage collagen is particularly
desirable. We describe an
advance in developing an immunoassay designed to measure pyridinoline
cross‑linked telopeptides from type II collagen in human urine,
serum, and synovial fluid. Synovial Fluid Markers of Osteoarthritis Kenneth D. Brandt, M.D. Development of potential disease‑modifying osteoarthritis (OA) drugs has prompted efforts to develop outcome measures of OA progression in humans. Although interest exists in various imaging procedures and arthroscopy, these techniques have not been validated or been proved suitable for use in clinical trials. Interest has grown, therefore, in biochemical/immunochemical tests for monitoring OA progression. Given the problems that exist in relating the serum concentration of a cartilage‑derived molecule to events in an index joint, it has been suggested that synovial fluid (SF) measurements may be more useful as markers of disease activity or severity in OA. However, unless factors that affect the kinetics of removal from the joint are taken into account, the SF concentration of these molecules cannot be a valid quantitative indicator of changes in articular cartilage metabolism. Although synovitis increases clearance of proteins from the joint space, we found that even after adjustment for the increased clearance rate in the OA knee, the synovial fluid concentration of sulfated glycosaminoglycans (most of which are derived from the cartilage) did not correlate with the severity of concurrent or subsequent cartilage damage. This emphasizes that the SF concentration of a cartilage‑derived marker is related not only to its clearance but also to other, more proximal variables, such as its rate of degradation, matrix permeability, and the rate of synthetic activity by chondrocytes in the OA cartilage. |