Autologous hematopoietic stem cell transplantation reverses skin fibrosis but does not change skin vessel density in patients with systemic sclerosis

Daikeler, Thomas; Kump, E; Stangel, Martin; Hügle, Thomas; Hij, A.; Haeuserman, P; Farge, Dominique

doi:10.1016/j.patbio.2015.07.006

Cited by 13 publications

(12 citation statements)

References 12 publications

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“…). Nine additional studies were identified by manual searching . We first removed 119 duplicates and then screened the abstracts before excluding an additional 226 studies because of irrelevance.…”

Section: Resultsmentioning

confidence: 99%

“…The characteristics of the included studies are summarized in Table and Table S3 (see Supporting Information). The study designs included four studies comparing autologous HSCT with CYC (two randomized studies, 89 patients), two nonrandomized controlled studies (20 patients) and 34 case series (247 patients) . There were three cohort studies derived from the EBMT registry .…”

Section: Resultsmentioning

confidence: 99%

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Efficacy and safety of autologous haematopoietic stem cell transplantation in systemic sclerosis: a systematic review of the literature

et al. 2018

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We aimed to assess the efficacy of autologous haematopoietic stem cell transplantation (HSCT) for skin sclerosis (SSc) and lung function in SSc. We performed a systematic literature review in the PubMed and Scopus databases from the earliest records to March 2016. We assessed study quality using the Cochrane tool for randomized studies, the Newcastle-Ottawa Scale for controlled cohort studies and an 18-item quality-appraisal checklist for case series. The primary outcome was the improvement of skin thickening using the modified Rodnan Skin Score (mRSS). The secondary outcome was efficacy on lung function, using diffusing capacity of the lungs for carbon monoxide and forced vital capacity (FVC). The safety of the procedure was evaluated. The literature search identified 431 citations. There were 38 studies involving a total of 344 patients who fulfilled our inclusion criteria. No meta-analysis was performed due to a high heterogeneity. There was a significant improvement in mRSS in the majority of the reports (P < 0·05), and the results were sustained for up to 8 years after autologous HSCT. The randomized studies and the four cohort studies each showed a slight but statistically significant improvement in FVC at 1 or 2 years. The treatment-related mortality calculated by pooling patients of 35 studies (336 patients with a follow-up up to 146 months) was 8·3% after autologous HSCT and 1% in cyclophosphamide-treated groups. Despite heterogeneity among the studies, we determined that autologous HSCT significantly improved cutaneous fibrosis and slightly improved FVC. Safety of autologous HSCT is acceptable given the severity of the disease. This systematic review was registered on PROSPERO, number CRD42016027951.

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Section: Resultsmentioning

confidence: 99%

Section: Resultsmentioning

confidence: 99%

Efficacy and safety of autologous haematopoietic stem cell transplantation in systemic sclerosis: a systematic review of the literature

et al. 2018

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“…Fleming et al have reported that high-dose immunosuppressive therapy and autologous stem-cell transplant increased capillary density in five of the seven subjects (10). However, these findings were not reproduced in a different group of patients undergoing similar therapy (20).…”

Section: Ec Apoptosismentioning

confidence: 98%

Pathogenesis of systemic sclerosis: recent insights of molecular and cellular mechanisms and therapeutic opportunities

Varga

Trojanowska

Kuwana

2017

Journal of Scleroderma and Related Disorders

277

251

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that results in irreversible scarring and organ failure. In other word, fibrosis is the end-result of a cascade of vascular and immune responses to environmental injury, and represents a failed tissue repair process. There have been advances in understanding of pathogenic processes that reflect the interplay between immune-inflammatory processes and vasculopathy and fibrosis. This review focuses on recent insights of functional consequences of SSc, highlighting three major characteristics of SSc; microvasculopathy, excessive fibrosis, and immune dysregulation. Microvasculopathy Although, widespread structural changes of the microvasculature are well documented in SSc patients, many aspects of SSc vasculopathy, including the nature of the injury and the fate of injured endothelial cells (ECs), remain poorly understood. Nevertheless, studies in recent years have brought a wealth of new information on the molecular and cellular mechanisms contributing to SSc vasculopathy. Diverse triggers have been implicated in inducing EC damage, including infection, immune-mediated cytotoxicity, anti-endothelial autoantibodies (AECAs), and ischemia-reperfusion injury. The resulting pathogenic changes in ECs may also vary and may include EC apoptosis, endothelial to mesenchymal transition (EndoMT), and other forms of EC activation. A bi-directional

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“…Studies on capillary density (the absolute number of nailfold capillaries) have been performed, in human normal and diseased tissues, including cancer, and showed significant correlations with severity of the pathological process and effects of therapies . However, the majority of studies have been performed on tissue specimens and implicate invasive and time‐consuming methods . On the other hand, direct microscopic analysis and quantification of skin capillaries have been recognized as noninvasive, safe, and fast methods …”

Section: Introductionmentioning

confidence: 99%

Automated assessment of absolute nailfold capillary number on videocapillaroscopic images: Proof of principle and validation in systemic sclerosis

et al. 2018

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Autologous hematopoietic stem cell transplantation reverses skin fibrosis but does not change skin vessel density in patients with systemic sclerosis

Cited by 13 publications

References 12 publications

Efficacy and safety of autologous haematopoietic stem cell transplantation in systemic sclerosis: a systematic review of the literature

Efficacy and safety of autologous haematopoietic stem cell transplantation in systemic sclerosis: a systematic review of the literature

Pathogenesis of systemic sclerosis: recent insights of molecular and cellular mechanisms and therapeutic opportunities

Automated assessment of absolute nailfold capillary number on videocapillaroscopic images: Proof of principle and validation in systemic sclerosis

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