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Scleroderma

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https://www.readbyqxmd.com/read/28340695/long-term-follow-up-after-autologous-adipose-derived-stromal-vascular-fraction-injection-into-fingers-in-systemic-sclerosis-patients
#1
A Daumas, J Magalon, E Jouve, R Truillet, D Casanova, L Giraudo, J Veran, A Benyamine, F Dignat-George, G Magalon, F Sabatier, B Granel
INTRODUCTION: Hand involvement confers a substantial handicap in work and daily activities in patients with Systemic sclerosis (SSc). Autologous adipose-derived stromal vascular fraction is as an easily accessible source of cells with regenerative effects. We previously performed a phase I open-label clinical trial (NTC01813279) assessing the safety of subcutaneous injection of autologous adipose-derived stromal vascular fraction. Six and 12-month data have been reported. As patients were followed in our medical centre, we report their longer-term outcome beyond the end of the trial...
January 2017: Current Research in Translational Medicine
https://www.readbyqxmd.com/read/28340090/predictive-value-of-european-scleroderma-group-activity-index-in-an-early-scleroderma-cohort
#2
Tatiana Nevskaya, Murray Baron, Janet E Pope
Objective.: To estimate the effect of disease activity, as measured by the European Scleroderma Research Group Activity Index (EScSG-AI), on the risk of subsequent organ damage in a large systemic sclerosis (SSc) cohort. Methods.: Of 421 SSc patients from the Canadian Scleroderma Research Group database with disease duration of ⩽ 3 years, 197 who had no evidence of end-stage organ damage initially and available 3 year follow-up were included. Disease activity was assessed by the EScSG-AI with two variability measures: the adjusted mean EScSG-AI (the area under the curve of the EScSG-AI over the observation period) and persistently active disease/flare...
March 17, 2017: Rheumatology
https://www.readbyqxmd.com/read/28337853/long-term-efficacy-and-tolerability-of-mycophenolate-mofetil-therapy-in-diffuse-scleroderma-skin-disease
#3
Daniel Boulos, Gene-Siew Ngian, Anton Rajadurai, Kathleen Elford, Wendy Stevens, Susanna Proudman, Claire Owen, Janet Roddy, Mandana Nikpour, Peter Youssef, Catherine Hill, Joanne Sahhar
OBJECTIVES: To assess the long-term efficacy and tolerability of mycophenolate mofetil (MMF) in patients with diffuse cutaneous systemic sclerosis (dcSSc). METHODS: Patients enrolled in the Australian Scleroderma Cohort study with dcSSc and baseline modified Rodnan skin score (mRSS) ≥ 12 who were treated for a minimum of 12 months with MMF for the primary indication of skin disease were included and their prospectively collected data retrieved. Change in mRSS, the proportion with a clinically significant improvement (reduction in mRSS ≥ 5 from baseline) and adverse effects due to therapy were determined...
March 24, 2017: International Journal of Rheumatic Diseases
https://www.readbyqxmd.com/read/28331982/autoantibodies-in-children-with-juvenile-dermatomyositis-a-single-centre-experience-from-north-west-india
#4
Altaf Hussain, Amit Rawat, Ankur Kumar Jindal, Anju Gupta, Surjit Singh
The objective of this study is to determine autoantibody profile in children with juvenile dermatomyositis (JDM). Children who were diagnosed with JDM (either recently diagnosed during the study period or follow-up patients) were included in the study. Autoantibodies were detected with commercially available Immunodot kit. Thirty patients were included in the study. Nine out of thirty patients (30%) were positive for one of the 12 autoantibodies tested. Anti-SRP antibody was most common antibody detected in 3 patients followed by anti-MDA-5 antibody in 2 patients; while anti-Jo1 antibody, anti-TIF1-γ antibody, anti-Mi-2 antibody, and anti-PM-Scl antibody were positive in 1 patient each...
March 22, 2017: Rheumatology International
https://www.readbyqxmd.com/read/28329476/pregnancy-associated-morphea-a-case-report-and-literature-review
#5
Anh Khoa Pham, Bhaskar Srivastava, April Deng
Morphea, also known as localized scleroderma, is arare fibrosing disorder of the skin, the pathogenesisof which is incompletely understood. It is thought,however, to involve interplay of genetic dispositionand triggering environmental factors, such asinfections and autoimmunity. Pregnancy as a potentialtrigger has only been reported in four cases. Herein,we present a patient who developed morphea of thebreasts during pregnancy, which rapidly resolvedwith a normal delivery. Our patient was distinct fromsome of the reported patients because her conditionwas tightly correlated with her pregnancy, as judgingby rapid resolution after delivery...
January 15, 2017: Dermatology Online Journal
https://www.readbyqxmd.com/read/28328178/clinical-characteristics-of-multifocal-osteonecrosis-in-korean-patients-with-rheumatic-disease
#6
Hye-Jin Jeong, Dam Kim, Soo-Kyung Cho, Yeesuk Kim, Sang-Cheol Bae, Yoon-Kyoung Sung
AIM: Osteonecrosis (ON), also known as avascular necrosis, is an important cause of physical disability in rheumatic disease. When this condition affects multiple structures, disability is increased. The purpose of this study was to describe the clinical characteristics of Korean patients with multifocal ON associated with rheumatic disease and to compare them with those of previously reported cases. METHODS: We reviewed the clinical characteristics of eight Korean patients with multifocal ON, defined by the involvement of three or more anatomic sites, associated with rheumatic disease in a single referral academic hospital, and compared them with those of 19 similar cases previously reported in the literature...
March 22, 2017: International Journal of Rheumatic Diseases
https://www.readbyqxmd.com/read/28319261/comparative-measurement-of-collagen-bundle-orientation-by-fourier-analysis-and-semiquantitative-evaluation-reliability-and-agreement-in-masson-s-trichrome-picrosirius-red-and-confocal-microscopy-techniques
#7
V Marcos-Garcés, M Harvat, P Molina Aguilar, A Ferrández Izquierdo, A Ruiz-Saurí
Measurement of collagen bundle orientation in histopathological samples is a widely used and useful technique in many research and clinical scenarios. Fourier analysis is the preferred method for performing this measurement, but the most appropriate staining and microscopy technique remains unclear. Some authors advocate the use of Haematoxylin-Eosin (H&E) and confocal microscopy, but there are no studies comparing this technique with other classical collagen stainings. In our study, 46 human skin samples were collected, processed for histological analysis and stained with Masson's trichrome, Picrosirius red and H&E...
March 20, 2017: Journal of Microscopy
https://www.readbyqxmd.com/read/28303554/imaging-of-the-oesophagus-beyond-cancer
#8
REVIEW
Thomas Marini, Amit Desai, Katherine Kaproth-Joslin, John Wandtke, Susan K Hobbs
Non-malignant oesophageal diseases are critical to recognize, but can be easily overlooked or misdiagnosed radiologically. In this paper, we cover the salient clinical features and imaging findings of non-malignant pathology of the oesophagus. We organize the many non-malignant diseases of the oesophagus into two major categories: luminal disorders and wall disorders. Luminal disorders include dilatation/narrowing (e.g. achalasia, scleroderma, and stricture) and foreign body impaction. Wall disorders include wall thickening (e...
March 17, 2017: Insights Into Imaging
https://www.readbyqxmd.com/read/28303481/morphea-and-eosinophilic-fasciitis-an-update
#9
REVIEW
Jorre S Mertens, Marieke M B Seyger, Rogier M Thurlings, Timothy R D J Radstake, Elke M G J de Jong
Morphea, also known as localized scleroderma, encompasses a group of idiopathic sclerotic skin diseases. The spectrum ranges from relatively mild phenotypes, which generally cause few problems besides local discomfort and visible disfigurement, to subtypes with severe complications such as joint contractures and limb length discrepancies. Eosinophilic fasciitis (EF, Shulman syndrome) is often regarded as belonging to the severe end of the morphea spectrum. The exact driving mechanisms behind morphea and EF pathogenesis remain to be elucidated...
March 16, 2017: American Journal of Clinical Dermatology
https://www.readbyqxmd.com/read/28303284/-generalized-circumscribed-scleroderma-with-blisters
#10
G Wagner, V Meyer, M M Sachse
The patient suffered from a 20-year course of generalized circumscribed scleroderma and presented with blisters in circumscribed areas of the affected skin. The development of subepidermal blisters has been described in all clinical forms of circumscribed scleroderma. Aetiology and pathogenesis of blister formation have not yet been clarified. An obstruction of the lymphatic vessels due to the present sclerosis is favoured. Treatment of bullous circumscribed scleroderma is considered to be difficult. Oral steroids, methotrexate, hydroxychloroquine and PUVA methods have been used with varying success...
March 16, 2017: Der Hautarzt; Zeitschrift Für Dermatologie, Venerologie, und Verwandte Gebiete
https://www.readbyqxmd.com/read/28301795/the-australian-scleroderma-interest-group-and-database-10-years-of-screening-to-save-lives
#11
Mandana Nikpour, Susanna Proudman, Kathleen Morrisroe, Joanne M Sahhar, Wendy Stevens
No abstract text is available yet for this article.
March 20, 2017: Medical Journal of Australia
https://www.readbyqxmd.com/read/28300892/scleromyxedema-clinical-diagnosis-and-autopsy-findings
#12
Ana Carolina Bulhões Sala, Paulo Rowilson Cunha, Clóvis Antônio Lopes Pinto, Célia Antônia Xavier de Moraes Alves, Ingrid Barreto Paiva, Ana Paula Vieira Araujo
Scleromyxedema is a rare chronic cutaneous mucinosis of unknown etiology. It is characterized by papular eruption and scleroderma with microscopic evidence of mucin deposition, fibroblast proliferation, and fibrosis. Most patients with scleromyxedema have monoclonal gammopathy and systemic manifestations resulting in significant morbidity and mortality. Several types of treatment have been reported with partial or inconsistent responses. Despite showing unpredictable evolution, systemic consequences of scleromyxedema and treatment side effects may result in death...
September 2016: Anais Brasileiros de Dermatologia
https://www.readbyqxmd.com/read/28299920/cervical-and-lumbar-paraspinal-calcinosis-in-systemic-sclerosis
#13
C K Tan, E Suresh
Calcinosis is a well-recognized manifestation of systemic sclerosis. Paraspinal or intraspinal calcinosis is rare, with reports of calcinosis involving the cervical, thoracic and lumbar separately, but not together. We now report a case of limited cutaneous scleroderma with extensive paraspinal calcinosis of the cervical and lumbar spine.
December 31, 2016: Reumatismo
https://www.readbyqxmd.com/read/28296274/relevance-of-clinical-and-autoantibody-profiles-in-systemic-sclerosis-among-thais
#14
Chingching Foocharoen, Piyakarn Watcharenwong, Sittichai Netwijitpan, Ajanee Mahakkanukrauh, Siraphop Suwannaroj, Ratanavadee Nanagara
OBJECTIVE: Autoantibody profiles in systemic sclerosis (SSc) and their relative clinical association vary between studies. The rate for being anti-topoisomerase-I (ATA) positive and the association with diffuse cutaneous the SSc subset (dcSSc) is higher among Thais than among Caucasians. The objective was to evaluate the relevance of clinical presentation, namely being positive for one or more autoantibodies among Thai SSc patients. METHOD: A retrospective, cohort study was performed among SSc patients over 18 years of age at Srinagarind Hospital, Khon Kaen University, Thailand, during January 2006 to December 2013...
March 10, 2017: International Journal of Rheumatic Diseases
https://www.readbyqxmd.com/read/28290895/-hemodynamic-predictors-of-clinical-deterioration-in-patients-with-pulmonary-arterial-hypertension-associated-with-systemic-scleroderma
#15
E V Nikolaeva, I A Kurmukov, N N Yudkina, S I Glukhova, A V Volkov
OBJECTIVE: to elucidate hemodynamic predictors of clinical deterioration (CD) in patients with pulmonary arterial hypertension (PAH) associated with systemic scleroderma (SSD). MATERIAL AND METHODS: We included into this study 48 patients with PAH-SSD consecutively admitted in 2004-2014. At inclusion all patients underwent right heart catheterization (RHC) and thereafter were under dynamic observation. CD deterioration was diagnosed in the presence of the following: >15% decline in 6-minute walk test distance; worsening of PAH functional class; intensification of symptoms of right ventricular failure; necessity in administration of parenteral diuretics...
October 2016: Kardiologiia
https://www.readbyqxmd.com/read/28285914/pharmacokinetic-optimitzation-of-ccg-203971-novel-inhibitors-of-the-rho-mrtf-srf-transcriptional-pathway-as-potential-antifibrotic-therapeutics-for-systemic-scleroderma
#16
Kim M Hutchings, Erika M Lisabeth, Walajapet Rajeswaran, Michael W Wilson, Roderick J Sorenson, Phillip L Campbell, Jeffrey H Ruth, Asif Amin, Pei-Suen Tsou, Jeffrey R Leipprandt, Samuel R Olson, Bo Wen, Ting Zhao, Duxin Sun, Dinesh Khanna, David A Fox, Richard R Neubig, Scott D Larsen
We recently reported the development of a novel inhibitor of Rho-mediated gene transcription (1, CCG-203971) that is efficacious in multiple animal models of acute fibrosis, including scleroderma, when given intraperitoneally. The modest in vivo potency and poor pharmacokinetics (PK) of this lead, however, make it unsuitable for long term efficacy studies. We therefore undertook a systematic medicinal chemistry effort to improve both the metabolic stability and the solubility of 1, resulting in the identification of two analogs achieving over 10-fold increases in plasma exposures in mice...
March 9, 2017: Bioorganic & Medicinal Chemistry Letters
https://www.readbyqxmd.com/read/28285783/histopathological-changes-in-morphea-and-their-clinical-correlates-results-from-the-morphea-in-adults-and-children-cohort-v
#17
Daniel Walker, Joseph S Susa, Sharif Currimbhoy, Heidi Jacobe
BACKGROUND: Histopathological features in morphea (localized scleroderma) and their clinical correlates are poorly described. OBJECTIVE: We sought to systematically describe histologic changes of morphea in a large, well-annotated cohort and determine the association between histopathology and clinical features. METHODS: This was a cross-sectional study of 83 patients enrolled in the Morphea in Adults and Children cohort. The main outcome measure was the association of microanatomical location and degree of sclerosis and inflammation seen on histologic samples with patient-reported symptoms and physician-based measures of severity...
March 9, 2017: Journal of the American Academy of Dermatology
https://www.readbyqxmd.com/read/28285170/novel-risk-factors-related-to-cancer-in-scleroderma
#18
REVIEW
David Bernal-Bello, Jaime García de Tena, Alfredo Guillén-Del Castillo, Albert Selva-O'Callaghan, Eduardo L Callejas-Moraga, Ana María Marín-Sánchez, Vicent Fonollosa-Pla, Carmen Pilar Simeón-Aznar
OBJECTIVE: Emerging data have shown an increased risk of malignancy among patients diagnosed with systemic sclerosis (SSc) so identification of risk factors linking both disorders might have prognostic implications. The aim of this study was to assess the clinical and treatment-related risk factors for cancer in a single-center cohort of patients with SSc. METHODS: Demographic, clinical, capillaroscopic, immunological and treatment-related data from 432 consecutive SSc patients were retrospectively analyzed...
March 8, 2017: Autoimmunity Reviews
https://www.readbyqxmd.com/read/28284324/readdressing-the-entity-of-exercise-pulmonary-arterial-hypertension
#19
REVIEW
Boris I Medarov, Sidharth Jogani, Johnathan Sun, Marc A Judson
Exercise pulmonary hypertension (EPH) indicates an abnormally elevated pulmonary artery pressure (PAP) during exercise. The physiological range of PAP during exercise remains poorly defined and, therefore, a universally accepted definition of EPH remains elusive. Nevertheless, previous data concerning the distribution of PAP in normal populations and more recent retrospective clinical data enhanced our ability to define EPH. EPH can impair exercise capacity and cause dyspnea. The underlying pathophysiology of the arterial form of EPH (EPAH) appears to be similar to that seen in resting pulmonary arterial hypertension (PAH), and EPAH individuals are at risk of developing resting PAH...
March 2017: Respiratory Medicine
https://www.readbyqxmd.com/read/28282243/differences-in-right-ventricular-functional-changes-during-treatment-between-systemic-sclerosis-associated-pulmonary-arterial-hypertension-and-idiopathic-pulmonary-arterial-hypertension
#20
Rahul G Argula, Abhijit Karwa, Abigail Lauer, David Gregg, Richard M Silver, Carol Feghali-Bostwick, Lynn M Schnapp, Kim Egbert, Bruce W Usher, Viswanathan Ramakrishnan, Paul M Hassoun, Charlie Strange
RATIONALE: Patients with scleroderma associated pulmonary arterial hypertension (SSc-PAH) continue to have an unacceptably high mortality despite the progress achieved with pulmonary arterial vasodilator therapies. OBJECTIVES: We sought to determine whether SSc-PAH is a clinically distinct pulmonary vascular disease phenotype when compared to IPAH, based on the progression of echocardiographic right ventricular (RV) dysfunction. METHODS: Retrospective analysis of echocardiographic data in 13 SSc-PAH and 11 IPAH patients was used to delineate the progression of RV dysfunction during single or combination pulmonary arterial vasodilator therapy...
March 10, 2017: Annals of the American Thoracic Society
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