ARTHRITIS & RHEUMATISM
Vol. 56, No. 9, September 2007, pp 3087–3095
DOI 10.1002/art.22835
© 2007, American College of Rheumatology
Adapted Versions of the Sharp/van der Heijde Score
Are Reliable and Valid for Assessment of
Radiographic Progression in Juvenile Idiopathic Arthritis
Angelo Ravelli,
1
Maka Ioseliani,
2
Ximena Norambuena,
2
Juliana Sato,
2
Angela Pistorio,
2
Federica Rossi,
2
Nicolino Ruperto,
2
Silvia Magni-Manzoni,
3
Nicola Ullmann,
2
and Alberto Martini
1
Objective. To develop adapted versions of the
Sharp/van der Heijde radiographic scoring system for
use in juvenile idiopathic arthritis (JIA), and to inves-
tigate their validity in JIA patients with polyarticular
disease.
Methods. The study group comprised 177 patients
with polyarticular JIA. Radiographs of the wrist/hand of
each patient were obtained at baseline (first observa-
tion) and then at 1, 3, 5, 7/8, and 10 years and were
assessed independently by 2 pediatric rheumatologists
according to different adaptations of the Sharp/van der
Heijde method. To facilitate score assignment, the ra-
diograph for each patient was compared with a bone
age–related standard. Validation procedures included
analysis of reliability, construct validity, and score
progression over time.
Results. Interobserver and intraobserver agree-
ment on longitudinal score values and score changes
was good for all of the adapted scoring versions (intra-
class correlation coefficient >0.85). Score changes over
time were moderately to strongly correlated with the
clinical indicators of long-term joint damage and with
the amount of long-term radiographic damage as mea-
sured with the carpo:metacarpal ratio, thereby demon-
strating good construct validity. A steady increase in
scores over time was observed, with joint space narrow-
ing being the most common form of damage throughout
the disease course. The inclusion of 5 new areas ap-
peared to increase the overall construct validity of
erosion scores.
Conclusion. Our results show that the adapted
versions of the Sharp/van der Heijde score are reliable
and valid for the assessment of radiographic progres-
sion in patients with JIA.
Juvenile idiopathic arthritis (JIA) is a chronic and
heterogeneous disease characterized by prolonged syno-
vial inflammation that may lead to destructive lesions of
joint structures (1). Because the prevention or retarda-
tion of joint changes is a major objective of treatment of
chronic arthritis, evaluation of radiographic joint dam-
age has become an important tool for assessing disease
severity and progression in patients with JIA. The
assessment of structural joint damage is considered the
gold standard of treatment efficacy studies in patients
with chronic arthritis (2) and is now required by the US
Food and Drug Administration to be used as a measure
of disease progression in clinical trials of potential
disease-modifying drugs (3). The evaluation of radio-
graphic progression has never been included in con-
trolled trials in JIA, reflecting primarily the paucity of
established radiographic scoring systems for use in the
pediatric age group. However, because new potent ther-
apeutic agents are now available for children with JIA
(4), there is a growing need for a reliable radiographic
assessment standard to investigate thoroughly the effec-
tiveness of these new agents.
Dr. Ioseliani is recipient of a bursary from the European
League Against Rheumatism. Drs. Norambuena and Sato are recipi-
ents of an Alpha Scholarship from the European Union (contract
AML/B7-311/970666/II-0246-FI).
1
Angelo Ravelli, MD, Alberto Martini, MD: Universita ` degli
Studi di Genoa, and IRCCS, Istituto G. Gaslini, Genoa, Italy;
2
Maka
Ioseliani, MD, Ximena Norambuena, MD, Juliana Sato, MD, Angela
Pistorio, MD, PhD, Federica Rossi, MD, Nicolino Ruperto, MD,
MPH, Nicola Ullmann, MD: IRCCS, Istituto G. Gaslini, Genoa, Italy;
3
Silvia Magni-Manzoni, MD: IRCCS, Policlinico S. Matteo, Pavia,
Italy.
Drs. Ioseliani and Norambuena contributed equally to this
work.
Address correspondence and reprint requests to Angelo
Ravelli, MD, Pediatria II, Istituto G. Gaslini, Largo G. Gaslini 5, 16147
Genoa, Italy. E-mail: angeloravelli@ospedale-gaslini.ge.it.
Submitted for publication December 29, 2006; accepted in
revised form May 16, 2007.
3087