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scleroderma and skin and treatment

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https://www.readbyqxmd.com/read/29121455/bench-to-bedside-and-back-again-finding-the-goldilocks-zone-within-the-scleroderma-universe
#1
EDITORIAL
Janet E Pope, Jason J Lee, Christopher P Denton
Systemic sclerosis (SSc) is a complex inflammatory connective tissue disease characterized by skin thickening, organ fibrosis and vasculopathy [1, 2]. The underlying disease mechanisms and pathophysiology are not fully understood. However, it appears to be, at least in part, driven by autoimmunity and inflammation associated with microvascular dysfunction ultimately resulting in excess extracellular matrix deposition in target organs [2-4]. Clinical heterogeneity coupled with our lack of understanding of the disease pathogenesis means that current treatment strategies are mostly organ based therapies using anti-rheumatic drugs that were originally approved for other indications [5]...
November 9, 2017: Arthritis & Rheumatology
https://www.readbyqxmd.com/read/29120857/the-safety-and-efficacy-of-light-emitting-diodes-based-ultraviolet-a1-phototherapy-in-bleomycin-induced-scleroderma-in-mice
#2
Diana Karpec, Romualdas Rudys, Laima Leonaviciene, Zygmunt Mackiewicz, Ruta Bradunaite, Gailute Kirdaite, Algirdas Venalis
PURPOSE: To define the efficacy and safety of narrowband ultraviolet A1 (UVA1) for the treatment of dermal fibrosis in bleomycin-induced mouse model of scleroderma. MATERIALS AND METHODS: 42 DBA/2 strain mice were included in the study: healthy mice and mice with established scleroderma, treated with high or medium dose of UVA1. Non-treated groups served as control. The equipment emitting 365±5nm UVA1 radiation was used in the study. The average cumulative doses were 1200J/cm(2) for high and 600J/cm(2) for medium dose course...
November 6, 2017: Advances in Medical Sciences
https://www.readbyqxmd.com/read/29104809/mycobacterium-intracellulare-infection-mimicking-progression-of-scleroderma
#3
Simon Krabbe, Merete Engelhart, Sören Thybo, Søren Jacobsen
This case report describes a patient with scleroderma who developed Mycobacterium intracellulare infection, which for more than a year mimicked worsening of her connective tissue disorder. The patient was diagnosed with scleroderma based on puffy fingers that developed into sclerodactyly, abnormal nail fold capillaries, interstitial lung disease, Raynaud's phenomenon, esophageal dysmotility, and positivity for rheumatoid factor and anti-SSA antibodies. She developed massive inflammatory changes of the cutis, the subcutis, and the muscle fasciae of the right leg, that after several failed attempts of immunosuppressive treatments were found to be caused by Mycobacterium intracellulare...
2017: Case Reports in Rheumatology
https://www.readbyqxmd.com/read/29093152/pomalidomide-in-patients-with-interstitial-lung-disease-due-to-systemic-sclerosis-a-phase-ii-multicenter-randomized-double-blind-placebo-controlled-parallel-group-study
#4
Vivien M Hsu, Christopher P Denton, Robyn T Domsic, Daniel E Furst, Maureen Rischmueller, Marina Stanislav, Virginia D Steen, Jörg H W Distler, Shimon Korish, Alyse Cooper, Suktae Choi, Peter H Schafer, Gerald Horan, Douglas R Hough
OBJECTIVE: To evaluate the safety and efficacy of pomalidomide (POM) on forced vital capacity (FVC), modified Rodnan skin score (mRSS), and gastrointestinal (GI) symptomatology over 52 weeks of treatment in patients with interstitial lung disease due to systemic sclerosis (SSc). METHODS: Twenty-three adult patients diagnosed with SSc were randomized 1:1 POM:placebo (PBO). RESULTS: Mean change at Week 52 from baseline in predicted FVC% -5.2 and -2...
November 1, 2017: Journal of Rheumatology
https://www.readbyqxmd.com/read/29065190/discovery-and-pharmacological-characterization-of-a-new-class-of-prolyl-trna-synthetase-inhibitor-for-anti-fibrosis-therapy
#5
Akira Shibata, Masako Kuno, Ryutaro Adachi, Yosuke Sato, Harumi Hattori, Atsushi Matsuda, Yuumi Okuzono, Keiko Igaki, Yusuke Tominari, Terufumi Takagi, Masato Yabuki, Masanori Okaniwa
Scleroderma has clinical characteristics including skin and other tissue fibrosis, but there is an unmet need for anti-fibrotic therapy. Halofuginone (HF) is a well-known anti-fibrosis agent in preclinical and clinical studies which exerts its effect via inhibition of TGF-β/Smad3 signaling pathway. Recently, prolyl-tRNA synthetase (PRS) was elucidated as a target protein for HF that binds to the proline binding site of the catalytic domain of PRS. Here, we characterized a new class of PRS inhibitor (T-3833261) that is carefully designed in a way that binds to the ATP site of the catalytic domain and does not disrupt binding of proline...
2017: PloS One
https://www.readbyqxmd.com/read/29058626/combined-platelet-rich-plasma-and-lipofilling-treatment-provides-great-improvement-in-facial-skin-induced-lesion-regeneration-for-scleroderma-patients
#6
Francesco Virzì, Paola Bianca, Alessandro Giammona, Tiziana Apuzzo, Simone Di Franco, Laura Rosa Mangiapane, Maria Luisa Colorito, Dario Catalano, Emanuela Scavo, Annalisa Nicotra, Antonina Benfante, Giuseppe Pistone, Valentina Caputo, Francesco Dieli, Roberto Pirrello, Giorgio Stassi
BACKGROUND: The use of stem cells, including mesenchymal stem cells (MSCs), for regenerative medicine is gaining interest for the clinical benefits so far obtained in patients. This study investigates the use of adipose autologous tissue in combination with platelet-rich plasma (PRP) to improve the clinical outcome of patients affected by systemic sclerosis (SSc). METHODS: Adipose-derived mesenchymal stem cells (AD-MSCs) and PRPs were purified from healthy donors and SSc patients...
October 23, 2017: Stem Cell Research & Therapy
https://www.readbyqxmd.com/read/28980909/tocilizumab-in-two-children-with-pansclerotic-morphoea-a-hopeful-therapy-for-refractory-cases
#7
Giorgia Martini, Simona Campus, Bernd Raffeiner, Gianluca Boscarol, Alessandra Meneghel, Francesco Zulian
Pansclerotic morphoea (PM) is a subtype of juvenile localised scleroderma characterised by severe course with generalised full-thickness skin involvement and possible growth and functional impairment. PM treatment comprises a combination of immunosuppressive agents such as corticosteroids, methotrexate, mycophenolate mofetil, PUVA and antithymocyte globulin and biological agents used in off-label. A possible role of IL-6 in the regulation of firoblast differentiation and stimulation of collagen synthesis has been suggested and in patients with systemic sclerosis (SSc) the treatment with tocilizumab (TCZ) was associated to improvement of skin thickness and joint motion...
September 2017: Clinical and Experimental Rheumatology
https://www.readbyqxmd.com/read/28980901/long-term-treatment-of-scleroderma-related-digital-ulcers-with-iloprost-a-cohort-study
#8
Michele Colaci, Federica Lumetti, Dilia Giuggioli, Serena Guiducci, Silvia Bellando-Randone, Ginevra Fiori, Marco Matucci-Cerinic, Clodoveo Ferri
OBJECTIVES: Raynaud's phenomenon and chronic/recurrent digital ulcers (DU) are main features of systemic sclerosis (SSc). Their treatment includes both systemic (i.e., iloprost) and local therapies. We report the therapeutic effects of iloprost in a cohort of SSc patients during a long-lasting follow-up period. METHODS: Fifty consecutive SSc patients (M/F 7/43, age at SSc diagnosis 43.5±12.7SD years) received iloprost infusions for 10±4.2SD years. Iloprost schedule consisted in monthly infusion at 0...
September 2017: Clinical and Experimental Rheumatology
https://www.readbyqxmd.com/read/28955239/searching-novel-therapeutic-targets-for-scleroderma-p2x7-receptor-is-up-regulated-and-promotes-a-fibrogenic-phenotype-in-systemic-sclerosis-fibroblasts
#9
Daniela Gentile, Pietro E Lazzerini, Alessandra Gamberucci, Mariarita Natale, Enrico Selvi, Francesca Vanni, Alessandra Alì, Paolo Taddeucci, Silvia Del-Ry, Manuela Cabiati, Veronica Della-Latta, David J Abraham, Maria A Morales, Rosella Fulceri, Franco Laghi-Pasini, Pier L Capecchi
Objectives: Systemic sclerosis (SSc) is a connective tissue disorder presenting fibrosis of the skin and internal organs, for which no effective treatments are currently available. Increasing evidence indicates that the P2X7 receptor (P2X7R), a nucleotide-gated ionotropic channel primarily involved in the inflammatory response, may also have a key role in the development of tissue fibrosis in different body districts. This study was aimed at investigating P2X7R expression and function in promoting a fibrogenic phenotype in dermal fibroblasts from SSc patients, also analyzing putative underlying mechanistic pathways...
2017: Frontiers in Pharmacology
https://www.readbyqxmd.com/read/28951708/morphea-in-middle-anatolia-turkey-a-5-year-single-center-experience
#10
Isil Bulur, Hilal Kaya Erdoğan, Tekden Karapınar, Zeynep Nurhan Saracoglu
INTRODUCTION: Morphea, also referred to as localized scleroderma, is a rare fibrosing skin disorder of undetermined cause. AIM: We report our single-center experience with morphea. MATERIAL AND METHODS: The study included 53 patients who were diagnosed with morphea by histopathology in our department between 2010 and 2015. Study data were collected retrospectively from the records of morphea patients. RESULTS: The study included 53 patients (38 women, 15 men), and median age at onset was 39...
August 2017: Postȩpy Dermatologii i Alergologii
https://www.readbyqxmd.com/read/28933138/assessment-of-treatment-effects-on-digital-ulcer-and-blood-perfusion-by-laser-speckle-contrast-analysis-in-a-patient-affected-by-systemic-sclerosis
#11
B Ruaro, S Paolino, C Pizzorni, M Cutolo, A Sulli
Laser speckle contrast analysis (LASCA) is a good tool to evaluate the variation in peripheral blood perfusion during long-term follow-up and is able to safely monitor digital ulcer evolution in scleroderma patients. It evaluates blood perfusion in different areas within the skin lesions and surrounding them during standard treatment.
September 21, 2017: Reumatismo
https://www.readbyqxmd.com/read/28899803/humanistic-and-cost-burden-of-systemic-sclerosis-a-review-of-the-literature
#12
REVIEW
Aryeh Fischer, Evelina Zimovetz, Caroline Ling, Dirk Esser, Nils Schoof
BACKGROUND: Systemic sclerosis (SSc), or systemic scleroderma, is a chronic multisystem autoimmune disease characterised by widespread vascular injury and progressive fibrosis of the skin and internal organs. Patients with SSc have decreased survival, with pulmonary involvement as the main cause of death. Current treatments for SSc manage a range of symptoms but not the cause of the disease. Our review describes the humanistic and cost burden of SSc. METHODS: A structured review of the literature was conducted, using predefined search strategies to search PubMed, Embase, and the Cochrane Library...
September 9, 2017: Autoimmunity Reviews
https://www.readbyqxmd.com/read/28885215/hyperspectral-imaging-as-a-novel-diagnostic-tool-in-microcirculation-of-wounds
#13
Georg Daeschlein, Inga Langner, Thomas Wild, Sebastian von Podewils, Claudia Sicher, Thomas Kiefer, Michael Jünger
Monitoring of perfusion is a cornerstone in surgery, phlebology and basic science to proof wound healing by interventions. In chronic wound management it is of utmost importance to realize and parametrize wound bed perfusion to verify actual, and plan further treatment by noninvasive diagnostics. Up to now monitoring is based on visual inspection of wounds as conventionally practiced over more than decades. The main problems of visual inspection are the lack of standardization and comparability because of interindividual variations...
September 4, 2017: Clinical Hemorheology and Microcirculation
https://www.readbyqxmd.com/read/28872977/growth-differentiation-factor-15-gdf-15-level-and-relation-to-clinical-manifestations-in-egyptian-systemic-sclerosis-patients-preliminary-data
#14
Sherif M Gamal, Fatema T Elgengehy, Asmaa Kamal, Samah A El Bakry, Elham Shabaan, Aliaa Elgendy, Iman H Bassyouni
AIM OF THE WORK: This study aims to assess Growth differentiation factor-15 (GDF-15) level in Scleroderma patients and its relation to disease manifestations. PATIENTS AND METHODS: This study included 55 scleroderma patients and 40 age and sex matched healthy volunteers. All patients were subjected to full history taking, thorough clinical examination, and laboratory investigations. GDF-15 serum levels were analyzed in patients and controls using human GDF-15 immunoassay Quantikine ELISA kit...
October 2017: Immunological Investigations
https://www.readbyqxmd.com/read/28870693/therapeutic-targeting-of-taz-and-yap-by-dimethyl-fumarate-in-systemic-sclerosis-fibrosis
#15
Tetsuo Toyama, Agnieszka P Looney, Brendon M Baker, Lukasz Stawski, Paul Haines, Robert Simms, Aleksander D Szymaniak, Xaralabos Varelas, Maria Trojanowska
Systemic sclerosis (scleroderma, SSc) is a devastating fibrotic disease with few treatment options. Fumaric acid esters, including dimethyl fumarate (DMF, Tecfidera®) have shown therapeutic effects in several disease models, prompting us to determine whether DMF is effective as a treatment for SSc dermal fibrosis. We found that DMF blocks the pro-fibrotic effects of TGFβ in SSc skin fibroblasts. Mechanistically, we found that DMF treatment reduced nuclear localization of TAZ and YAP proteins via inhibition of PI3-K/Akt1 pathway...
September 1, 2017: Journal of Investigative Dermatology
https://www.readbyqxmd.com/read/28726006/microrna-21-in-skin-fibrosis-potential-for-diagnosis-and-treatment
#16
REVIEW
Yan Li, Juan Zhang, Yuying Lei, Lechun Lyu, Ruiling Zuo, Ting Chen
Skin fibrosis is a common pathological process characterized by fibroblast proliferation and excessive deposition of extracellular matrix. However, the pathogenesis of the disease is still not clear. Previous studies have shown that microRNA-21 may play pivotal roles in the regulation of a variety of skin fibrosis, including keloid, scleroderma, and hypertrophic scar. In this review, we outline the structure, expression, and regulation of microRNA-21 and its role in fibrotic skin diseases. In future, it may be useful as a prognostic or diagnostic marker...
December 2017: Molecular Diagnosis & Therapy
https://www.readbyqxmd.com/read/28712039/cutaneous-manifestations-of-scleroderma-and-scleroderma-like-disorders-a-comprehensive-review
#17
REVIEW
Caterina Ferreli, Giulia Gasparini, Aurora Parodi, Emanuele Cozzani, Franco Rongioletti, Laura Atzori
Scleroderma refers to an autoimmune connective tissue fibrosing disease, including three different subsets: localized scleroderma, limited cutaneous systemic sclerosis, and diffuse cutaneous systemic sclerosis with divergent patterns of organ involvement, autoantibody profiles, management, and prognostic implications. Although systemic sclerosis is considered the disease prototype that causes cutaneous sclerosis, there are many other conditions that can mimic and be confused with SSc. They can be classified into immune-mediated/inflammatory, immune-mediated/inflammatory with abnormal deposit (mucinoses), genetic, drug-induced and toxic, metabolic, panniculitis/vascular, and (para)neoplastic disorders according to clinico-pathological and pathogenetic correlations...
July 16, 2017: Clinical Reviews in Allergy & Immunology
https://www.readbyqxmd.com/read/28703365/phototherapy-in-systemic-sclerosis-review
#18
REVIEW
Suteeraporn Chaowattanapanit, Charoen Choonhakarn, Chingching Foocharoen, Narachai Julanon
Systemic scleroderma-also known as systemic sclerosis (SSc)-is a chronic systemic connective tissue disease characterized by collagen deposition in cutaneous and internal organs, leading to skin sclerosis and multiple organ fibrosis. The pathogenesis is complex and remains poorly understood. Treatment is based on organ involvement and requires a multidisciplinary approach. Skin sclerosis can cause disability, leading to decreasing quality of life. Various systemic antifibrotic therapies have been used; however, most have unsatisfactory results...
July 13, 2017: Photodermatology, Photoimmunology & Photomedicine
https://www.readbyqxmd.com/read/28691464/parry-romberg-syndrome-and-linear-facial-scleroderma-management-in-pediatric-population
#19
E Segna, V Pucciarelli, G A Beltramini, C Sforza, F J Silvestre, A B Giannì, A Baj
Among all different types of cutaneous scleroderma, Parry Romberg syndrome and linear scleroderma “en coup de sabre” typically involve the visage. Gradual degeneration of the tissues, from the skin up to the bone, is the stigmata of the diseases and the range of clinical manifestations is wide. They typically start during childhood and slowly progress before stabilizing. Considering the gravity of the associated deformity and its impact on facial function and appearance, we strongly advocate a prompt intervention that however must be tailored on paediatric patients...
April 2017: Journal of Biological Regulators and Homeostatic Agents
https://www.readbyqxmd.com/read/28562527/successful-treatment-of-infliximab-in-a-patient-with-scleroderma-a-case-report
#20
Ting Li, Yaoyang Liu, Huji Xu
RATIONALE: Systemic Scleroderma (SSc) is a rare connective tissue disease clinically characterized by cutaneous sclerosis and variable systemic involvement. No drug is currently available to effectively reverse the fibrotic process in SSc. Previous reports have suggested that the tumor necrosis factor (TNF) antagonists could be useful for the treatment of fibrotic disorders. However, TNFα has long been considered as an antifibrotic cytokine. Whether TNF antagonist is effective for SSc patients needs to be tested...
June 2017: Medicine (Baltimore)
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