S-160
1
Clinica Medica, Dipartimento di Medicina
Interna, Ospedali Riuniti, Ancona, Italy;
2
Clinica di Reumatologia, Dipartimento
di Patologia e Medicina Sperimentale e
Clinica, Università di Udine, Udine, Italy;
3
Dipartimento di Scienze Cliniche e
Molecolari, Università Politecnica delle
Marche, Ancona, Italy;
4
Istituto di Anatomia Patologica,
Dipartimento di Ricerche Mediche e
Morfologiche Università di Udine,
Udine, Italy.
*These authors contributed equally to
this work.
Paolo Fraticelli, MD
Salvatore De Vita, MD
Nicoletta Franzolini, MD
Silvia Svegliati, PhD
Caterina A. Scott, MD
Cecilia Tonnini, PhD
Tatiana Spadoni, PhD
Barbara Gabrielli, MD
Giovanni Pomponio, MD
Gianluca Moroncini, MD, PhD
Armando Gabrielli, MD
Please address correspondence to:
Dr Armando Gabrielli,
Dipartimento di Scienze
Cliniche e Molecolari,
Università Politecnica delle Marche,
Via Tronto 10,
60020 Ancona, Italy.
E-mail: a.gabrielli@univpm.it
Received on June 9, 2015; accepted in
revised form on July 1, 2015.
Clin Exp Rheumatol 2015; 33 (Suppl. 91):
S160-S167.
© Copyright CliniCal and
ExpErimEntal rhEumatology 2015.
Key words: systemic sclerosis,
B cells, autoimmune disease
Funding: this work was supported in part
by the Società Italiana di Reumatologia,
Associazione Italiana per la Lotta alla
Sclerodermia, Ministero Italiano per
l’Università e la Ricerca Scientiica and
by a special grant from the Fondazione
Cariverona e Fondazione di Medicina
Molecolare e Terapia Cellulare, Ancona,
Italy.
Competing interests: none declared.
ABSTRACT
Objective. There is evidence that B
lymphocytes play a role in the patho-
genesis of systemic sclerosis (scleroder-
ma). Stimulatory autoantibodies target-
ing and activating normal human ibro-
blasts in vitro have been demonstrated
in sera from scleroderma patients.
Rituximab is a monoclonal antibody
which selectively targets and depletes
CD20
+
B lymphocytes. We investigated
the biological effects of rituximab in six
patients affected by scleroderma with
severe skin involvement.
Methods. Six patients with severe skin
ibrosis, unresponsive to immunosup-
pressive treatment, were treated with
375 mg/m
2
per week of intravenous
rituximab for a total of four doses. Se-
rum stimulatory autoantibodies to the
PDGF receptor were detected. Fibro-
blast activation was evaluated in ibro-
blasts grown from skin biopsies per-
formed at baseline and at months 3 and
6 post-treatment. The modiied Rodnan’s
skin score, health assessment question-
naire (HAQ) and visual analogic scale
(VAS) for global wellness and B lym-
phocyte count were performed monthly.
Results. A signiicant reduction of an-
ti-PDGF receptor autoantibodies was
observed in the serum of all patients
3 months after treatment. Fibroblasts
showed a signiicant downregulation
of type I collagen gene expression and
of the intracellular signalling trig-
gered by anti-PDGFR autoantibod-
ies. A decrease of the skin score and
an improvement of disability indexes
matched with the in vitro results. A sin-
gle course of rituximab reduced scle-
roderma ibroblast activation in vitro
and the serum levels of anti-PDGFR
stimulatory autoantibodies.
Conclusion. These data provide fur-
ther evidence of B-cell involvement
in the pathogenesis of scleroderma.
Targeting B cells may be a promising
treatment for scleroderma patients, and
controlled clinical trials are warranted.
Introduction
Systemic sclerosis (SSc) is a connec-
tive tissue disease characterised by
increased production of extracellular
matrix by ibroblasts, both in the skin
and in visceral organs. The aetiology is
unknown, and key pathogenetic events
include immune system activation, en-
dothelial cell dysfunction and ibroblast
activation (1-3).
Although current therapies can control
end organ damage (4), no disease-mod-
ifying agent able to induce complete re-
mission of the disease is available yet,
probably due to the poor knowledge
of the mechanisms implicated in the
pathophysiology of the disease (5).
It has been shown that SSc patients are
characterised by activation of humoral
immunity. Whitield et al. using DNA
microarrays have revealed up-regula-
tion of genes related to B cells in SSc
skin lesions (6). Furthermore, memory
B cells have been found to be activated
in scleroderma patients and showed
enhanced ability to produce IgG in
vitro under well deined experimental
conditions (7). Increased serum levels
of BAFF or B cell-activating factor,
a potent B cell survival factor, were
detected in an unselected SSc popula-
tion and found to correlate positively
with the extent of skin ibrosis (8). The
activation of B cells may lead to in-
creased production of IL-6 and TGF-β
which induce ibrosis (9) and IL-10 and
IL-6 which drive a Th2-dominant im-
mune response (10, 11) involved in the
mechanisms of autoantibody produc-
tion (12). The description of several
autoantibodies directed against non-nu-
Reduced type I collagen gene expression by skin ibroblasts
of patients with systemic sclerosis after one treatment course
with rituximab
P. Fraticelli
1
, S. De Vita
2
, N. Franzolini
2
, S. Svegliati
3
, C.A. Scott
4
, C. Tonnini
3
,
T. Spadoni
3
, B. Gabrielli
3
, G. Pomponio
1
, G. Moroncini
1,3
, A. Gabrielli
1,3