Resolution of SLE-related soft-tissue calcification following haematopoietic stem cell transplantation

Mandelbrot, Didier A.; Santos, Peter W.; Burt, Richard K.; Oyama, Yu; Block, Geoffrey A.; Ahya, Shubhada N.; Rosa, Robert M.; Traynor, Ann E.

doi:10.1093/ndt/gfn036

Cited by 19 publications

(24 citation statements)

References 42 publications

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“…13,14 The deposition of calcification can be idiopathic, dystrophic, or due to calciphylaxis, and while the pathophysiology is unclear, it is thought to relate to circulating chronic inflammatory mediators. [15][16][17][18][19][20][21][22][23][24][25][26][27] Case reports describe both focal and extensive systemic deposition of calcification in patients with both rheumatoid arthritis and SLE, with calciphylaxis described as a rare complication of both diseases. [16][17][18][19][20][21][22][23] SLE can lead to CKD, a well-known complication 22 ; however, CKD was not independently associated with trochlear calcifications.…”

Section: Discussionmentioning

confidence: 99%

“…Widespread soft-tissue calcification in chronic inflammatory autoimmune disorders such as Sjögren syndrome, SLE, and rheumatoid arthritis has been described. [20][21][22][23][25][26][27] Very few case reports describe focal calcifications within the soft tissues of the head and neck, with the exception of retropharyngeal calcific tendinitis and calcification of the longus colli muscle in a patient with rheumatoid arthritis. 28 On the basis of the results of this investigation, it appears that in addition to calcification within soft tissues previously described, trochlear calcifications may also be seen as an associated finding in patients with autoimmune conditions.…”

Section: Discussionmentioning

confidence: 99%

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Clinical Significance of Trochlear Calcifications in the Orbit

Buch¹,

Nadgir²,

Tannenbaum³

et al. 2013

AJNR Am J Neuroradiol

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BACKGROUND AND PURPOSE:Trochlear calcification is frequently seen on CT scans that include the orbits, but prior studies linking this finding to diabetes mellitus were based on selected populations by using relatively thick sections. We assessed the relationship between trochlear calcification and diabetes mellitus and other chronic medical conditions in an unrestricted population by using thin-section CT.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Clinical Significance of Trochlear Calcifications in the Orbit

Buch¹,

Nadgir²,

Tannenbaum³

et al. 2013

AJNR Am J Neuroradiol

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“…Resolution of soft-tissue calcification has also been reported [41]. Interestingly, recent posttransplant analyses have revealed CD8 + Treg are significantly increased in SLE patients posttransplant and these are proposed to play a central role in restoration of tolerance post-transplant [42].…”

Section: Haematopoietic Stem Cell Transplantation For Autoimmune Diseasementioning

confidence: 89%

Induction of antigen-specific tolerance through hematopoietic stem cell-mediated gene therapy: The future for therapy of autoimmune disease?

Coleman

Steptoe

2012

Autoimmunity Reviews

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“…Autologous HSCT also proved effective in antiphospholipid syndrome: 10 of 22 patients discontinued anticoagulation therapy, 78% of whom remained without further thrombotic events [ 27 ]. Interestingly, soft tissue calcifications resolved after autologous HSCT [ 28 ].…”

Section: Clinical Datamentioning

confidence: 99%

Stem cell transplantation for rheumatic autoimmune diseases

Hügle

Laar

2008

Arthritis Res Ther

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Immunoablative therapy and hematopoietic stem cell transplantation (HSCT) is an intensive treatment modality aimed at 'resetting' the dysregulated immune system of a patient with immunoablative therapy and allow outgrowth of a nonautogressive immune system from reinfused hematopoietic stem cells, either from the patient (autologous HSCT) or a healthy donor (allogeneic HSCT). HSCT has been shown to induce profound alterations of the immune system affecting B and T cells, monocytes, and natural killer and dendritic cells, resulting in elimination of autoantibodyproducing plasma cells and in induction of regulatory T cells. Most of the available data have been collected through retrospective cohort analyses of autologous HSCT, case series, and translational studies in patients with refractory autoimmune diseases. Long-term and marked improvements of disease activity have been observed, notably in systemic sclerosis, systemic lupus erythematosus, and juvenile idiopathic arthritis, and treatment-related morbidity and mortality have improved due to better patient selection and modifications of transplant regimens. Treatment-related mortality has decreased to approximately 7%. Prospective, randomised, controlled clinical trials are ongoing or planned in systemic sclerosis, systemic lupus erythematosus, and several nonrheumatological conditions. IntroductionData from nearly 1,000 patients with refractory autoimmune disease (AD) treated with hematopoietic stem cell transplantation (HSCT) have been collected by the European Group for Blood and Marrow Transplantation/European League Against Rheumatism (EBMT/EULAR) Working Party for Autoimmune Diseases in the past 10 years (Tables 1 and 2; Riccardo Saccardi, chair of the EBMT/EULAR Working Party, personal communication) [1][2][3][4]. The advent of this multistep treatment modality ( Figure 1) followed clinical observations of remissions of AD in patients who were transplanted for concomitant hemato-oncological conditions [5]. The immunological principles were subsequently confirmed through mechanistic studies in animal models of AD [6]. HSCT in AD was widely pioneered in the 1990s at a time when few biologicals were available to treat AD and when refractory progressive disease posed a major challenge. The introduction of effective biologicals for the treatment of rheumatoid arthritis (RA), juvenile idiopathic arthritis (JIA), ankylosing spondylitis, and systemic lupus erythematosus (SLE) has reduced the demand for intensive and toxic treatments such as HSCT. Nevertheless, clinicians still face cases of severe and life-threatening AD such as progressive systemic sclerosis (SSc), vasculitis, or SLE, refractory to conventional medication and biologicals, for whom HSCT may be a therapeutic option. Whereas biologics target one cell type or soluble cytokine only, HSCT affects all immune effector cells involved in AD, notably B and T lymphocytes, monocytes, natural killer (NK) cells, and dendritic cells (DCs). In contrast to conventional immunosuppressive medication and b...

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Resolution of SLE-related soft-tissue calcification following haematopoietic stem cell transplantation

Abstract: The successful treatment of advanced calcium deposition by aggressive immune ablation underscores the contribution of SLE-mediated inflammation to soft-tissue calcification syndromes.

Cited by 19 publications

References 42 publications

Clinical Significance of Trochlear Calcifications in the Orbit

Clinical Significance of Trochlear Calcifications in the Orbit

Induction of antigen-specific tolerance through hematopoietic stem cell-mediated gene therapy: The future for therapy of autoimmune disease?

Stem cell transplantation for rheumatic autoimmune diseases

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