104 The Journal of Rheumatology 2011; 38:1; doi:10.3899/jrheum.100191
Personal non-commercial use only. The Journal of Rheumatology Copyright © 2011. All rights reserved.
Systemic Vasculitis in Patients with Hepatitis C Virus
Infection with and without Detectable Mixed
Cryoglobulinemia
BENJAMIN TERRIER, DAMIEN SÈNE, AGNÈS DECHARTRES, DAVID SAADOUN, NICOLAS ORTONNE,
PHILIPPE ROUVIER, LUCILE MUSSET, MATTHIEU RESCHE RIGON, THIERRY MAISONOBE,
and PATRICE CACOUB
ABSTRACT. Objective. To describe hepatitis C virus (HCV)-related systemic vasculitis in patients without
detectable mixed cryoglobulinemia (MC) and to compare them to typical cases of HCV-MC
vasculitis.
Methods. Twelve HCV RNA+ patients with histologically proven vasculitis in the absence of
detectable MC (cases) were retrospectively compared with 48 HCV RNA+ patients with MC vas-
culitis (controls). Each case was matched with 4 controls for age and sex.
Results. The main epidemiological and virologic features were similar between cases and controls.
No clinical difference was found, except for lower rates of arthralgias (33% vs 71%; p = 0.02) and
purpura (50% vs 83%; p = 0.03) in cases. Cases showed higher mean serum C3 (1.17 ± 0.21 vs 0.93
± 0.23 g/l; p = 0.01) and median C4 levels (0.25 vs 0.04 g/l; p < 0.001), lower median serum IgM
levels (0.6 vs 1.9 g/l; p < 0.001), and lower rates of rheumatoid factor positivity (8% vs 82%; p <
0.001) than controls. The main histologic features were similar between cases and controls.
Immunofluorescence analysis of skin biopsy from 1 case revealed perivascular deposits of C3 and
IgA. After treatment, overall clinical response of vasculitis (75% vs 83%) and sustained virological
response (40% vs 64%; p = 0.3) were similar between cases and controls, except for higher complete
clinical response (42% vs 73%; p = 0.05) in controls.
Conclusion. HCV-related systemic vasculitis may occur in the absence of detectable MC. Our find-
ings suggest that such vasculitis probably results from immune complex-mediated mechanisms, and
that the therapeutic management of such vasculitis should be similar to that of HCV-MC vasculitis.
(First Release Oct 15 2010; J Rheumatol 2011;38:104–10; doi:10.3899/jrheum.100191)
Key Indexing Terms:
HEPATITIS C VIRUS VASCULITIS MIXED CRYOGLOBULINEMIA
POLYARTERITIS NODOSA ANTIVIRAL THERAPY
From the Department of Internal Medicine, CNRS UMR 7211,
Department of Pathology, Department of Immunology, Department of
Neuropathology, Assistance Publique-Hôpitaux de Paris (APHP), Groupe
Hospitalier Pitié-Salpétrière, Université Pierre et Marie Curie, Paris;
Department of Biostatistics and Medical Data Processing, Hôpital St
Louis, Paris; and Department of Pathology, APHP, Hôpital Henri
Mondor, Créteil, France.
B. Terrier, MD, Department of Internal Medicine, CNRS UMR 7211;
D. Sène, MD, PhD, Department of Internal Medicine; A. Dechartres, MD,
Department of Biostatistics and Medical Data Processing, Hôpital St
Louis; D. Saadoun, MD, PhD, Department of Internal Medicine, CNRS
UMR 7211; N. Ortonne, MD, Department of Pathology, APHP, Hôpital
Henri Mondor; P. Rouvier, MD, Department of Pathology; L. Musset,
MD, Department of Immunology, APHP Groupe Hospitalier
Pitié-Salpétrière; M. Resche-Rigon, MD, PhD, Department of Biostatistics
and Medical Data Processing, Hôpital St Louis; T. Maisonobe, MD,
Department of Neuropathology, APHP Groupe Hospitalier
Pitié-Salpétrière; P. Cacoub, MD, PhD, Department of Internal Medicine,
CNRS UMR 7211.
Address correspondence to Prof. P. Cacoub, Department of Internal
Medicine, APHP Groupe Hospitalier Pitié-Salpétrière, 47 boulevard de
l’Hôpital, 75013 Paris, France. E-mail: patrice.cacoub@psl.aphp.fr
Accepted for publication August 17, 2010.
Systemic vasculitis comprises a group of disorders defined
by inflammation of the blood vessel walls, which are classi-
fied according to their clinical and histologic features and
the size of the predominantly affected vessels. Various path-
ogenic mechanisms have been implicated in the induction of
vasculitis: (1) cell-mediated inflammation; (2) immune
complex-mediated inflammation such as mixed cryoglobu-
linemia (MC) vasculitis and polyarteritis nodosa (PAN)
associated with hepatitis B virus infection; and (3) autoanti-
body-mediated inflammation such as antineutrophil cyto-
plasmic antibody (ANCA)-associated vasculitis
1,2,3
.
Shortly after the discovery of hepatitis C virus (HCV) in
1989, there was evidence that more than 80% of MC cases
were associated with HCV infection
4,5,6
. In addition, among
patients with HCV-related MC (HCV-MC), the prevalence
of symptomatic vasculitis ranges from < 1% to 10% accord-
ing to geographic distribution, with < 5% generally found in
larger prospective studies
7,8,9
. MC is a systemic vasculitis
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