Systemic sclerosisTreatment of Systemic Sclerosis-Associated Calcinosis: A Case Report of Rituximab-Induced Regression of CREST-Related Calcinosis and Review of the Literature
Section snippets
Methods
We performed an extensive Internet search (Medline) using the keywords calcinosis, calcification, scleroderma, systemic sclerosis, and treatment in the following combinations: systemic sclerosis (or scleroderma) and calcinosis (or calcification) and treatment. The limits used were (1) English language, (2) human subjects, and (3) published from 1981 and onward. The search identified 392 articles; following duplicate removal, 182 articles remained and were screened for eligibility. One hundred
Literature Review
In this section, we review all the available pharmacologic (summarized in Table 1) and nonpharmacologic treatment options for SSc-associated calcinosis.
Discussion
Treatment of SSc-associated calcinosis has been so far disappointing. Even though several modalities have been tried, none has definitely shown a beneficial effect. We present herein the first case of RTX administration in limited SSc; all patients recruited so far in the studies assessing the efficacy of RTX in SSc belonged to the diffuse form of the disease. The rationale for the use of RTX in SSc is based on experimental evidence from animal models, indicating that B cells may be actively
References (61)
- et al.
Is there a role for B-cell depletion as therapy for scleroderma?A case report and review of the literature
Semin Arthritis Rheum
(2010) - et al.
The presence of gamma-carboxyglutamic acid in the proteins associated with ectopic calcification
Biochem Biophys Res Commun
(1976) - et al.
Treatment of calcinosis universalis with low-dose warfarin
Am J Med
(1987) - et al.
Failure of warfarin in treatment of calcinosis universalis
Am J Med
(1988) - et al.
Post-translational modifications regulate matrix Gla protein function: importance for inhibition of vascular smooth muscle cell calcification
J Thromb Haemost
(2007) - et al.
Relation of oral anticoagulation to cardiac valvular and coronary calcium assessed by multislice spiral computed tomography
Am J Cardiol
(2005) - et al.
Oral anticoagulant treatment: friend or foe in cardiovascular disease?
Blood
(2004) - et al.
Treatment of cutaneous calcinosis in CREST syndrome by extracorporeal shock wave lithotripsy
J Am Acad Dermatol
(2005) - et al.
Complete resolution of universal calcinosis in a patient with juvenile dermatomyositis using pamidronate
Joint Bone Spine
(2010) - et al.
B Lymphocyte signaling established by the CD19/CD22 loop regulates autoimmunity in the tight-skin mouse
Am J Pathol
(2004)
B-lymphocyte depletion reduces skin fibrosis and autoimmunity in the tight-skin mouse model for systemic sclerosis
Am J Pathol
CD19 regulates skin and lung fibrosis via Toll-like receptor signaling in a model of bleomycin-induced scleroderma
Am J Pathol
Rituximab-induced B cell depletion in autoimmune diseases: potential effects on T cells
Clin Immunol
Clinical and laboratory associations of anticentromere antibody in patients with progressive systemic sclerosis
Arthritis Rheum
Experience with rituximab in scleroderma: results from a 1-year, proof-of-principle study
Rheumatology (Oxford)
Successful treatment of resistant scleroderma-associated interstitial lung disease with rituximab
Rheumatology (Oxford)
Successful treatment of steroid and cyclophosphamide-resistant diffuse scleroderma-associated interstitial lung disease with rituximab
Rheumatol Int
Cyclophosphamide-refractory scleroderma-associated interstitial lung disease: remarkable clinical and radiological response to a single course of rituximab combined with high-dose corticosteroids
Ther Adv Respir Dis
Rituximab in diffuse cutaneous systemic sclerosis: an open-label clinical and histopathological study
Ann Rheum Dis
B cell depletion in diffuse progressive systemic sclerosis: safety, skin score modification and IL-6 modulation in an up to thirty-six months follow-up open-label trial
Arthritis Res Ther
B cell depletion with rituximab in patients with diffuse cutaneous systemic sclerosis
Arthritis Rheum
Gamma-carboxyglutamate excretion and calcinosis in juvenile dermatomyositis
Arthritis Rheum
Effect of warfarin sodium therapy on excretion of 4-carboxy-L-glutamic acid in scleroderma, dermatomyositis, and myositis ossificans progressiva
Arthritis Rheum
Low dose warfarin treatment for calcinosis in patients with systemic sclerosis
Ann Rheum Dis
The effects of warfarin on calcinosis in a patient with systemic sclerosis
J Rheumatol
Spontaneous calcification of arteries and cartilage in mice lacking matrix GLA protein
Nature
Extracellular matrix mineralization is regulated locally; different roles of two gla-containing proteins
J Cell Biol
Mutations in the gene encoding the human matrix Gla protein cause Keutel syndrome
Nat Genet
Matrix GLA protein and BMP-2 regulate osteoinduction in calcifying vascular cells
J Cell Biochem
Warfarin causes rapid calcification of the elastic lamellae in rat arteries and heart valves
Arterioscler Thromb Vasc Biol
Cited by (0)
The authors have no conflicts of interest to disclose.
- 1
These authors contributed equally to this article.