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2. Safety and efficacy of subcutaneous tocilizumab in adults with systemic sclerosis (faSScinate): a phase 2, randomised, controlled trial

Author

Khanna, Dinesh, Denton, Christopher P, Jahreis, Angelika, van Laar, Jacob M, Frech, Tracy M, Anderson, Marina E, Baron, Murray, Chung, Lorinda, Fierlbeck, Gerhard, Lakshminarayanan, Santhanam, Allanore, Yannick, Pope, Janet E, Riemekasten, Gabriela, Steen, Virginia, Müller-Ladner, Ulf, Lafyatis, Robert, Stifano, Giuseppina, Spotswood, Helen, Chen-Harris, Haiyin, Dziadek, Sebastian, Morimoto, Alyssa, Sornasse, Thierry, Siegel, Jeffrey, and Furst, Daniel E

Published
2016
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4. Safety and efficacy of subcutaneous tocilizumab in systemic sclerosis: results from the open-label period of a phase II randomised controlled trial (faSScinate)

Author

Khanna, Dinesh, Denton, Christopher P, Lin, Celia J F, van Laar, Jacob M, Frech, Tracy M, Anderson, Marina E, Baron, Murray, Chung, Lorinda, Fierlbeck, Gerhard, Lakshminarayanan, Santhanam, Allanore, Yannick, Pope, Janet E, Riemekasten, Gabriela, Steen, Virginia, Müller-Ladner, Ulf, Spotswood, Helen, Burke, Laura, Siegel, Jeffrey, Jahreis, Angelika, and Furst, Daniel E

Published
2018
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6. Prior elicitation of the efficacy and tolerability of Methotrexate and Mycophenolate Mofetil in Juvenile Localised Scleroderma

Author

Desai, Yasin, primary, Jaki, Thomas, additional, Beresford, Michael W, additional, Burnett, Thomas, additional, Eleftheriou, Despina, additional, Jacobe, Heidi, additional, Leone, Valentina, additional, Li, Suzanne, additional, Mozgunov, Pavel, additional, Ramanan, Athimalaipet V, additional, Torok, Kathryn S, additional, Anderson, Marina E, additional, Anton, Jordi, additional, Avcin, Tadej, additional, Felton, Jessie, additional, Foeldvari, Ivan, additional, Laguda, Bisola, additional, McErlane, Flora, additional, Shaw, Lindsay, additional, Zulian, Francesco, additional, and Pain, Clare E, additional

Published
2021
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7. Connective tissue disease related interstitial lung diseases and idiopathic pulmonary fibrosis: provisional core sets of domains and instruments for use in clinical trials

Author

Saketkoo, Lesley Ann, Mittoo, Shikha, Huscher, Dörte, Khanna, Dinesh, Dellaripa, Paul F, Distler, Oliver, Flaherty, Kevin R, Frankel, Sid, Oddis, Chester V, Denton, Christopher P, Fischer, Aryeh, Kowal-Bielecka, Otylia M, LeSage, Daphne, Merkel, Peter A, Phillips, Kristine, Pittrow, David, Swigris, Jeffrey, Antoniou, Katerina, Baughman, Robert P, Castelino, Flavia V, Christmann, Romy B, Christopher-Stine, Lisa, Collard, Harold R, Cottin, Vincent, Danoff, Sonye, Highland, Kristin B, Hummers, Laura, Shah, Ami A, Kim, Dong Soon, Lynch, David A, Miller, Frederick W, Proudman, Susanna M, Richeldi, Luca, Ryu, Jay H, Sandorfi, Nora, Sarver, Catherine, Wells, Athol U, Strand, Vibeke, Matteson, Eric L, Brown, Kevin K, Seibold, James R, Aggarwal, Rohit, Ainslie, Gillian, Alkassab, Firas, Allanore, Yannick, Descartes, Paris, Anderson, Marina E, Andonopoulos, Andrew P, Antin-Ozerkis, Danielle, Arrobas, Ana, Ascherman, Dana P, Assassi, Shervin, Baron, Murray, Bathon, Joan M, Behr, Juergen, Beretta, Lorenzo, Bingham, Clifton O, III, Binnie, Matthew, Birring, Surinder S, Boin, Francesco, Bongartz, Tim, Bourdin, Arnaud, Bouros, Demosthenes, Brasington, Richard, Bresser, Paul, Buch, Maya H, Burge, P Sherwood, Carmona, Loreto, Carreira, Patricia E, Carvalho, Carlos RR, Catoggio, Luis J, Chan, Kevin M, Chapman, Jeffrey, Chatterjee, Soumya, Chua, Felix, Chung, Lorinda, Conron, Matthew, Corte, Tamera, Cosgrove, Gregory, Costabel, Ulrich, Cox, Gerard, Crestani, Bruno, Crofford, Leslie J, Csuka, Mary E, Curbelo, Pablo, László, Czirják, Daniil, Zoe, DʼArsigny, Christine L, Davis, Gerald S, de Andrade, Joao A, De Vuyst, Paul, Dempsey, Owen J, Derk, Chris T, Distler, Jörg, Dixon, William G, Downey, Gregory, Doyle, Mittie K, Drent, Marjolein, Durairaj, Lakshmi, Emery, Paul, Espinoza, Luis R, Farge, Dominique, Fathi, Maryam, Fell, Charlene D, Fessler, Barri J, Fitzgerald, John E, Fox, George A, Foeldvari, Ivan, Frech, Tracy M, Freitas, Sara, Furst, Daniel E, Gabrielli, Armando, García-Vicuña, Rosario, Georgiev, Ognian B, Gerbino, Anthony, Gillisen, Adrian, Gladman, Dafna D, Glassberg, Marilyn, Gochuico, Bernadette R, Gogali, Athena, Goh, Nicole S, Goldberg, Avram, Goldberg, Hilary J, Gourley, Mark F, Griffing, Leroy, Grutters, Jan C, Gunnarsson, Ragnar, Hachulla, Eric, Hall, Frances C, Harari, Sergio, Herrick, Ariane L, Herzog, Erica L, Hesselstrand, Roger, Hirani, Nikhil, Hodgson, Ulla, Hollingsworth, Helen M, Homer, Robert J, Hoyles, Rachel K, Hsu, Vivien M, Hubbard, Richard B, Hunzelmann, Nicolas, Isasi, Maria Eloisa, Isasi, Elida Susana, Sergio A Jimenez, Jacobsen Soren, Johnson, Sindhu R, Jones, Christine H, Kahaleh, Bashar, Kairalla, Ronaldo A, Kalluri, Meena, Kalra, Sanjay, Kaner, Robert J, Kinder, Brent W, Klingsberg, Ross C, Kokosi, Maria, RJ Kolb, Martin, Kur-Zalewska, Joanna, Kuwana, Masataka, Lake, Fiona R, Lally, Edward V, Lasky, Joseph A, Laurindo, Ileda M, Able, Lawrence, Lee, Peter, Leonard, Colm T, Lien, Dale C, Limper, Andrew H, Liossis, Stamatis-Nick C, Lohr, Kristine M, Loyd, James E, Lundberg, Ingrid E, Mageto, Yolanda N, Maher, Toby M, Mahmud, Tafazzul H, Manganas, Helene, Marie, Isabelle, Marras, Theodore K, Antônio Baddini Martinez, José, Martinez, Fernando J, Mathieu, Alessandro, Matucci-Cerinic, Marco, Mayes, Maureen D, McKown, Kevin M, Medsger, Thomas A, Jr., Meehan, Richard T, Cristina, Mendes Ana, Meyer, Keith C, Millar, Ann B, Moğulkoç, Nesrin, Molitor, Jerry A, Morais, António, Luc Mouthon, Portugal, Müller, Veronika, Müller-Quernheim, Joachim, Nadashkevich, Oleg, Nador, Roland, Nash, Peter, Nathan, Steven D, Navarro, Carmen, Neves, Sofia, Noth, Imre, Nunes, Hilario, Olson, Amy L., Opitz, Christian F, Padilla, Maria, Pappas, Dimitrios, Parfrey, Helen, Pego-Reigosa, José M, AC Pereira, Carlos, Perez, Rafael, Pope, Janet E, Porter, Joanna C., Renzoni, Elisabeth A, Riemekasten, Gabriela, Riley, David J, Rischmueller, Maureen, Rodriguez-Reyna, Tatiana S, Rojas-Serrano, Romam, Jesse, Rosen, Glenn D, Rossman, Milton, Rothfield, Naomi, Sahn, Steven A, Sanduzzi, Alessandro, Scholand, Mary Beth, Selman, Moises, Senécal, Jean-Luc, Seo, Philip, Silver, Richard M, Solomon, Joshua J, Steen, Virginia, Stevens, Wendy, Strange, Charlie, Sussman, Robert, Sutton, Evelyn D, Sweiss, Nadera J, Tornling, Göran, Tzelepis, George E, Undurraga, Alvaro, Vacca, Allessandra, Vancheri, Carlo, Varga, Janos, Veale, Douglas J, Volkov, Suncica, Walker, Ulrich A, Wencel, Mark, Wesselius, Lewis J, Wickremasinghe, Melissa, Wilcox, Pearce, Wilsher, Margaret L, Wollheim, Frank A, Wuyts, Wim A, Yung, Gordon, Zanon, Pietro, Zappala, Christopher J, Groshong, Steve D, Leslie, Kevin O, Myers, Jeffrey L, Padera, Robert F, R Desai, Sujal, Goldin, Jonathan, Kazerooni, Ella A, Klein, Jeffrey S, Lynch, David A, and Keen, Kevin J

Published
2014
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8. Prior elicitation of the efficacy and tolerability of Methotrexate and Mycophenolate Mofetil in Juvenile Localised Scleroderma

Author

Desai, Yasin, Jaki, Thomas, Beresford, Michael W, Burnett, Thomas, Eleftheriou, Despina, Jacobe, Heidi, Leone, Valentina, Li, Suzanne, Mozgunov, Pavel, Ramanan, Athimalaipet V, Torok, Kathryn S, Anderson, Marina E, Anton, Jordi, Avcin, Tadej, Felton, Jessie, Foeldvari, Ivan, Laguda, Bisola, McErlane, Flora, Shaw, Lindsay, Zulian, Francesco, Pain, Clare E, Desai, Yasin, Jaki, Thomas, Beresford, Michael W, Burnett, Thomas, Eleftheriou, Despina, Jacobe, Heidi, Leone, Valentina, Li, Suzanne, Mozgunov, Pavel, Ramanan, Athimalaipet V, Torok, Kathryn S, Anderson, Marina E, Anton, Jordi, Avcin, Tadej, Felton, Jessie, Foeldvari, Ivan, Laguda, Bisola, McErlane, Flora, Shaw, Lindsay, Zulian, Francesco, and Pain, Clare E

Abstract

BackgroundEvidence is lacking for safe and effective treatments for juvenile localised scleroderma (JLS). Methotrexate (MTX) is commonly used first line and mycophenolate mofetil (MMF) second line, despite a limited evidence base. A head to head trial of these two medications would provide data on relative efficacy and tolerability. However, a frequentist approach is difficult to deliver in JLS, because of the numbers needed to sufficiently power a trial. A Bayesian approach could be considered.MethodsAn international consensus meeting was convened including an elicitation exercise where opinion was sought on the relative efficacy and tolerability of MTX compared to MMF to produce prior distributions for a future Bayesian trial. Secondary aims were to achieve consensus agreement on critical aspects of a future trial.ResultsAn international group of 12 clinical experts participated. Opinion suggested superior efficacy and tolerability of MMF compared to MTX; where most likely value of efficacy of MMF was 0.70 (95% confidence interval (CI) 0.34-0.90) and of MTX was 0.68 (95% CI 0.41-0.8). The most likely value of tolerability of MMF was 0.77 (95% CI 0.3-0.94) and of MTX was 0.62 (95% CI 0.32-0.84). The wider CI for MMF highlights that experts were less sure about relative efficacy and tolerability of MMF compared to MTX. Despite using a Bayesian approach, power calculations still produced a total sample size of 240 participants, reflecting the uncertainty amongst experts about the performance of MMF.ConclusionsKey factors have been defined regarding the design of a future Bayesian approach clinical trial including elicitation of prior opinion of the efficacy and tolerability of MTX and MMF in JLS. Combining further efficacy data on MTX and MMF with prior opinion could potentially reduce the pre-trial uncertainty so that, when combined with smaller trial sample sizes a compelling evidence base is available.

Published
2021

15. Patterns and predictors of skin score change in early diffuse systemic sclerosis from the European Scleroderma Observational Study

Author

Herrick, Ariane, Peytrignet, Sebastien, Lunt, Mark, Pan, Xiaoyan, Dinsdale, Graham, Brown, Edith, Czirják, László, Distler, Jörg H.W., Distler, Oliver, Fligelstone, Kim, Gregory, William J, Ochiel, Rachel, Vonk, Madelon C, Ancuta, Codrina, Ong, Voon H, Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Øyvind, Jobanputra, Paresh, Jordan, Alison C, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C, Agard, Christian, Anderson, Marina E, Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya, Chung, Lorinda, Damjanov, Nemanja, Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Soren, MacGregor, Alexander J, McHugh, Neil, Müller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E, Hachulla, Eric, Hamilton, Jennifer, Inanç, Murat, McLaren, John S, van Laar, Jacob M., Pathare, Sanjay, Proudman, Susannah, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J, Grange, Claire, Trad, Georges-Selim, and Denton, Christopher P.

Published
2018
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16. Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study

Author

Peytrignet, Sébastien, Denton, Christopher P, Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Pan, Xiaoyan, Brown, Edith, Czirják, László, Distler, Jörg H W, Distler, Oliver, Fligelstone, Kim, Gregory, William J, Ochiel, Rachel, Vonk, Madelon, Ancuta, Codrina, Ong, Voon H, Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Øyvind, Jordan, Alison C, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C, Agard, Christian, Anderson, Marina E, Diot, Elisabeth, Madhok, Rajan, et al, and University of Zurich

Subjects
early diffuse cutaneous systemic sclerosis, disability, 2745 Rheumatology, 10051 Rheumatology Clinic and Institute of Physical Medicine, 2736 Pharmacology (medical), 610 Medicine & health, fatigue, pain, hand function
Published
2018
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17. A MULTICENTRE RELIABILITY AND VALIDITY STUDY OF LASER SPECKLE CONTRAST IMAGING AND THERMOGRAPHY IN PATIENTS WITH RAYNAUD'S PHENOMENON SECONDARY TO SYSTEMIC SCLEROSIS

Author

Murray, Andrea K, Manning, Joanne, Moore, Tonia, Wilkinson, Jack, Marjanovic, Elizabeth, Leggett, Sarah, Dinsdale, Graham, Roberts, Christopher, Allen, John, Anderson, Marina E, Britton, Jason, Buch, Maya, Del Galdo, Francesco, Denton, Christopher P, Drayton, Tracey, Furlong, Anita, Griffiths, Bridget, Hall, Frances, Hart, Darren, Howell, Kevin, MacDonald, Audrey, McHugh, Neil J, Pauling, John, Shipley, Jacqueline, and Herrick, Ariane

Published
2017

18. Nailfold capillaroscopy—how many fingers should be examined to detect abnormality?

Author

Dinsdale, Graham, primary, Roberts, Chris, additional, Moore, Tonia, additional, Manning, Joanne, additional, Berks, Michael, additional, Allen, John, additional, Anderson, Marina E, additional, Cutolo, Maurizio, additional, Hesselstrand, Roger, additional, Howell, Kevin, additional, Pizzorni, Carmen, additional, Smith, Vanessa, additional, Sulli, Alberto, additional, Wildt, Marie, additional, Taylor, Christopher, additional, Murray, Andrea, additional, and Herrick, Ariane L, additional

Published
2018
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19. Patient-reported outcome instruments for assessing Raynaud’s phenomenon in systemic sclerosis: A SCTC vascular working group report

Author

Pauling, John D, primary, Frech, Tracy M, additional, Hughes, Michael, additional, Gordon, Jessica K, additional, Domsic, Robyn T, additional, Anderson, Marina E, additional, Ingegnoli, Francesca, additional, McHugh, Neil J, additional, Johnson, Sindhu R, additional, Hudson, Marie, additional, Boin, Francesco, additional, Ong, Voon H, additional, Matucci-Cerinic, Marco, additional, Altorok, Nezam, additional, Scolnik, Marina, additional, Nikpour, Mandana, additional, Shah, Ankoor, additional, Pope, Janet E, additional, Khanna, Dinesh, additional, and Herrick, Ariane L, additional

Published
2018
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20. A Multicenter Study of the Validity and Reliability of Responses to Hand Cold Challenge as Measured by Laser Speckle Contrast Imaging and Thermography

Author

Wilkinson, Jack D., primary, Leggett, Sarah A., additional, Marjanovic, Elizabeth J., additional, Moore, Tonia L., additional, Allen, John, additional, Anderson, Marina E., additional, Britton, Jason, additional, Buch, Maya H., additional, Del Galdo, Francesco, additional, Denton, Christopher P., additional, Dinsdale, Graham, additional, Griffiths, Bridgett, additional, Hall, Frances, additional, Howell, Kevin, additional, MacDonald, Audrey, additional, McHugh, Neil J., additional, Manning, Joanne B., additional, Pauling, John D., additional, Roberts, Christopher, additional, Shipley, Jacqueline A., additional, Herrick, Ariane L., additional, and Murray, Andrea K., additional

Published
2018
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22. Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis:the European Scleroderma Observational Study

Author

Peytrignet, Sébastien, Denton, Christopher P, Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Pan, Xiaoyan, Brown, Edith, Czirják, László, Distler, Jörg H W, Distler, Oliver, Fligelstone, Kim, Gregory, William J, Ochiel, Rachel, Vonk, Madelon, Ancuta, Codrina, Ong, Voon H, Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Øyvind, Jordan, Alison C, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C, Agard, Christian, Anderson, Marina E, Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H, Chung, Lorinda, Damjanov, Nemanja, Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Søren, MacGregor, Alexander J, McHugh, Neil, Müller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E, Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, Inanç, Murat, McLaren, John S, van Laar, Jacob M, Pathare, Sanjay, Proudman, Susanna, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J, Grange, Claire, Trad, Georges-Selim, Herrick, Ariane L, Peytrignet, Sébastien, Denton, Christopher P, Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Pan, Xiaoyan, Brown, Edith, Czirják, László, Distler, Jörg H W, Distler, Oliver, Fligelstone, Kim, Gregory, William J, Ochiel, Rachel, Vonk, Madelon, Ancuta, Codrina, Ong, Voon H, Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Øyvind, Jordan, Alison C, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C, Agard, Christian, Anderson, Marina E, Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H, Chung, Lorinda, Damjanov, Nemanja, Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Søren, MacGregor, Alexander J, McHugh, Neil, Müller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E, Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, Inanç, Murat, McLaren, John S, van Laar, Jacob M, Pathare, Sanjay, Proudman, Susanna, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J, Grange, Claire, Trad, Georges-Selim, and Herrick, Ariane L

Published
2018

23. Patterns and predictors of skin score change in early diffuse systemic sclerosis from the European Scleroderma Observational Study

Author

Herrick, Ariane L., Peytrignet, Sebastien, Lunt, Mark, Pan, Xiaoyan, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan J., Dinsdale, Graham, Brown, Edith, Czirjak, Laszlo, Distler, Joerg H. W., Distler, Oliver, Fligelstone, Kim, Gregory, William J., Ochiel, Rachel, Vonk, Madelon C., Ancuta, Codrina, Ong, Voon H., Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Oyvind, Jobanputra, Paresh, Jordan, Alison C., Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C., Agard, Christian, Anderson, Marina E., Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H., Chung, Lorinda, Damjanov, Nemanja S., Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Soren, MacGregor, Alexander J., McHugh, Neil, Mueller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E., Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, Inanc, Murat, McLaren, John S., van Laar, Jacob M., Pathare, Sanjay, Proudman, Susanna M., Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J., Grange, Claire, Trad, Georges-Selim, Denton, Christopher P., Herrick, Ariane L., Peytrignet, Sebastien, Lunt, Mark, Pan, Xiaoyan, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan J., Dinsdale, Graham, Brown, Edith, Czirjak, Laszlo, Distler, Joerg H. W., Distler, Oliver, Fligelstone, Kim, Gregory, William J., Ochiel, Rachel, Vonk, Madelon C., Ancuta, Codrina, Ong, Voon H., Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Oyvind, Jobanputra, Paresh, Jordan, Alison C., Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C., Agard, Christian, Anderson, Marina E., Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H., Chung, Lorinda, Damjanov, Nemanja S., Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Soren, MacGregor, Alexander J., McHugh, Neil, Mueller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E., Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, Inanc, Murat, McLaren, John S., van Laar, Jacob M., Pathare, Sanjay, Proudman, Susanna M., Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J., Grange, Claire, Trad, Georges-Selim, and Denton, Christopher P.

Abstract

Objectives Our aim was to use the opportunity provided by the European Scleroderma Observational Study to (1) identify and describe those patients with early diffuse cutaneous systemic sclerosis (dcSSc) with progressive skin thickness, and (2) derive prediction models for progression over 12 months, to inform future randomised controlled trials (RCTs). Methods The modified Rodnan skin score (mRSS) was recorded every 3months in 326 patients. Progressors' were defined as those experiencing a 5-unit and 25% increase in mRSS score over 12 months (3 months). Logistic models were fitted to predict progression and, using receiver operating characteristic (ROC) curves, were compared on the basis of the area under curve (AUC), accuracy and positive predictive value (PPV). Results 66 patients (22.5%) progressed, 227 (77.5%) did not (33 could not have their status assessed due to insufficient data). Progressors had shorter disease duration (median 8.1 vs 12.6 months, P=0.001) and lower mRSS (median 19 vs 21 units, P=0.030) than non-progressors. Skin score was highest, and peaked earliest, in the anti-RNA polymerase III (Pol3+) subgroup (n=50). A first predictive model (including mRSS, duration of skin thickening and their interaction) had an accuracy of 60.9%, AUC of 0.666 and PPV of 33.8%. By adding a variable for Pol3 positivity, the model reached an accuracy of 71%, AUC of 0.711 and PPV of 41%. Conclusions Two prediction models for progressive skin thickening were derived, for use both in clinical practice and for cohort enrichment in RCTs. These models will inform recruitment into the many clinical trials of dcSSc projected for the coming years. Trial registration number NCT02339441.

Published
2018

24. Patterns and predictors of skin score change in early diffuse systemic sclerosis from the European Scleroderma Observational Study

Author

Herrick, Ariane L, Peytrignet, Sebastien, Lunt, Mark, Pan, Xiaoyan, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan J, Dinsdale, Graham, Brown, Edith, Czirják, László, Distler, Jörg H W, Distler, Oliver, Fligelstone, Kim, Gregory, William J, Ochiel, Rachel, Vonk, Madelon C, Ancuţa, Codrina, Ong, Voon H, Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Øyvind, Jobanputra, Paresh, Jordan, Alison C, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C, Agard, Christian, Anderson, Marina E, Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H, Chung, Lorinda, Damjanov, Nemanja S, Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Søren, MacGregor, Alexander J, McHugh, Neil, Müller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E, Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, İnanç, Murat, McLaren, John S, van Laar, Jacob M, Pathare, Sanjay, Proudman, Susanna M, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J, Grange, Claire, Trad, Georges-Selim, Denton, Christopher P, Herrick, Ariane L, Peytrignet, Sebastien, Lunt, Mark, Pan, Xiaoyan, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan J, Dinsdale, Graham, Brown, Edith, Czirják, László, Distler, Jörg H W, Distler, Oliver, Fligelstone, Kim, Gregory, William J, Ochiel, Rachel, Vonk, Madelon C, Ancuţa, Codrina, Ong, Voon H, Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Øyvind, Jobanputra, Paresh, Jordan, Alison C, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C, Agard, Christian, Anderson, Marina E, Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H, Chung, Lorinda, Damjanov, Nemanja S, Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Søren, MacGregor, Alexander J, McHugh, Neil, Müller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E, Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, İnanç, Murat, McLaren, John S, van Laar, Jacob M, Pathare, Sanjay, Proudman, Susanna M, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J, Grange, Claire, Trad, Georges-Selim, and Denton, Christopher P

Abstract

OBJECTIVES: Our aim was to use the opportunity provided by the European Scleroderma Observational Study to (1) identify and describe those patients with early diffuse cutaneous systemic sclerosis (dcSSc) with progressive skin thickness, and (2) derive prediction models for progression over 12 months, to inform future randomised controlled trials (RCTs).METHODS: The modified Rodnan skin score (mRSS) was recorded every 3 months in 326 patients. 'Progressors' were defined as those experiencing a 5-unit and 25% increase in mRSS score over 12 months (±3 months). Logistic models were fitted to predict progression and, using receiver operating characteristic (ROC) curves, were compared on the basis of the area under curve (AUC), accuracy and positive predictive value (PPV).RESULTS: 66 patients (22.5%) progressed, 227 (77.5%) did not (33 could not have their status assessed due to insufficient data). Progressors had shorter disease duration (median 8.1 vs 12.6 months, P=0.001) and lower mRSS (median 19 vs 21 units, P=0.030) than non-progressors. Skin score was highest, and peaked earliest, in the anti-RNA polymerase III (Pol3+) subgroup (n=50). A first predictive model (including mRSS, duration of skin thickening and their interaction) had an accuracy of 60.9%, AUC of 0.666 and PPV of 33.8%. By adding a variable for Pol3 positivity, the model reached an accuracy of 71%, AUC of 0.711 and PPV of 41%.CONCLUSIONS: Two prediction models for progressive skin thickening were derived, for use both in clinical practice and for cohort enrichment in RCTs. These models will inform recruitment into the many clinical trials of dcSSc projected for the coming years.TRIAL REGISTRATION NUMBER: NCT02339441.

Published
2018

25. Treatment outcome in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study (ESOS)

Author

Herrick, Ariane L, Pan, Xiaoyan, Peytrignet, Sébastien, Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Brown, Edith, Czirják, László, Distler, Jörg H W, Distler, Oliver, Fligelstone, Kim, Gregory, William J, Ochiel, Rachel, Vonk, Madelon, Ancuţa, Codrina, Ong, Voon H, Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Øyvind, Jordan, Alison C, Jobanputra, Paresh, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C, Agard, Christian, Anderson, Marina E, Diot, Elisabeth, et al, University of Zurich, and Herrick, Ariane L

Subjects
2403 Immunology, 1300 General Biochemistry, Genetics and Molecular Biology, 2745 Rheumatology, 10051 Rheumatology Clinic and Institute of Physical Medicine, 2723 Immunology and Allergy, 610 Medicine & health
Published
2017
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26. Patterns and predictors of skin score change in early diffuse systemic sclerosis from the European Scleroderma Observational Study

Author

Herrick, Ariane L, primary, Peytrignet, Sebastien, additional, Lunt, Mark, additional, Pan, Xiaoyan, additional, Hesselstrand, Roger, additional, Mouthon, Luc, additional, Silman, Alan J, additional, Dinsdale, Graham, additional, Brown, Edith, additional, Czirják, László, additional, Distler, Jörg H W, additional, Distler, Oliver, additional, Fligelstone, Kim, additional, Gregory, William J, additional, Ochiel, Rachel, additional, Vonk, Madelon C, additional, Ancuţa, Codrina, additional, Ong, Voon H, additional, Farge, Dominique, additional, Hudson, Marie, additional, Matucci-Cerinic, Marco, additional, Balbir-Gurman, Alexandra, additional, Midtvedt, Øyvind, additional, Jobanputra, Paresh, additional, Jordan, Alison C, additional, Stevens, Wendy, additional, Moinzadeh, Pia, additional, Hall, Frances C, additional, Agard, Christian, additional, Anderson, Marina E, additional, Diot, Elisabeth, additional, Madhok, Rajan, additional, Akil, Mohammed, additional, Buch, Maya H, additional, Chung, Lorinda, additional, Damjanov, Nemanja S, additional, Gunawardena, Harsha, additional, Lanyon, Peter, additional, Ahmad, Yasmeen, additional, Chakravarty, Kuntal, additional, Jacobsen, Søren, additional, MacGregor, Alexander J, additional, McHugh, Neil, additional, Müller-Ladner, Ulf, additional, Riemekasten, Gabriela, additional, Becker, Michael, additional, Roddy, Janet, additional, Carreira, Patricia E, additional, Fauchais, Anne Laure, additional, Hachulla, Eric, additional, Hamilton, Jennifer, additional, İnanç, Murat, additional, McLaren, John S, additional, van Laar, Jacob M, additional, Pathare, Sanjay, additional, Proudman, Susanna M, additional, Rudin, Anna, additional, Sahhar, Joanne, additional, Coppere, Brigitte, additional, Serratrice, Christine, additional, Sheeran, Tom, additional, Veale, Douglas J, additional, Grange, Claire, additional, Trad, Georges-Selim, additional, and Denton, Christopher P, additional

Published
2018
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27. Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study

Author

Peytrignet, Sébastien, primary, Denton, Christopher P, additional, Lunt, Mark, additional, Hesselstrand, Roger, additional, Mouthon, Luc, additional, Silman, Alan, additional, Pan, Xiaoyan, additional, Brown, Edith, additional, Czirják, László, additional, Distler, Jörg H W, additional, Distler, Oliver, additional, Fligelstone, Kim, additional, Gregory, William J, additional, Ochiel, Rachel, additional, Vonk, Madelon, additional, Ancuţa, Codrina, additional, Ong, Voon H, additional, Farge, Dominique, additional, Hudson, Marie, additional, Matucci-Cerinic, Marco, additional, Balbir-Gurman, Alexandra, additional, Midtvedt, Øyvind, additional, Jordan, Alison C, additional, Stevens, Wendy, additional, Moinzadeh, Pia, additional, Hall, Frances C, additional, Agard, Christian, additional, Anderson, Marina E, additional, Diot, Elisabeth, additional, Madhok, Rajan, additional, Akil, Mohammed, additional, Buch, Maya H, additional, Chung, Lorinda, additional, Damjanov, Nemanja, additional, Gunawardena, Harsha, additional, Lanyon, Peter, additional, Ahmad, Yasmeen, additional, Chakravarty, Kuntal, additional, Jacobsen, Søren, additional, MacGregor, Alexander J, additional, McHugh, Neil, additional, Müller-Ladner, Ulf, additional, Riemekasten, Gabriela, additional, Becker, Michael, additional, Roddy, Janet, additional, Carreira, Patricia E, additional, Fauchais, Anne Laure, additional, Hachulla, Eric, additional, Hamilton, Jennifer, additional, İnanç, Murat, additional, McLaren, John S, additional, van Laar, Jacob M, additional, Pathare, Sanjay, additional, Proudman, Susanna, additional, Rudin, Anna, additional, Sahhar, Joanne, additional, Coppere, Brigitte, additional, Serratrice, Christine, additional, Sheeran, Tom, additional, Veale, Douglas J, additional, Grange, Claire, additional, Trad, Georges-Selim, additional, and Herrick, Ariane L, additional

Published
2017
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28. Patient acceptable symptom state in scleroderma: results from the tocilizumab compared with placebo trial in active diffuse cutaneous systemic sclerosis

Author

Arnold, Michael B, primary, Khanna, Dinesh, additional, Denton, Christopher P, additional, van Laar, Jacob M, additional, Frech, Tracy M, additional, Anderson, Marina E, additional, Baron, Murray, additional, Chung, Lorinda, additional, Fierlbeck, Gerhard, additional, Lakshminarayanan, Santhanam, additional, Allanore, Yannick, additional, Riemekasten, Gabriela, additional, Steen, Virginia, additional, Müller-Ladner, Ulf, additional, Spotswood, Helen, additional, Burke, Laura, additional, Siegel, Jeffrey, additional, Jahreis, Angelika, additional, Furst, Daniel E, additional, and Pope, Janet E, additional

Published
2017
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29. Safety and efficacy of subcutaneous tocilizumab in systemic sclerosis: results from the open-label period of a phase II randomised controlled trial (faSScinate)

Author

Khanna, Dinesh, primary, Denton, Christopher P, additional, Lin, Celia J F, additional, van Laar, Jacob M, additional, Frech, Tracy M, additional, Anderson, Marina E, additional, Baron, Murray, additional, Chung, Lorinda, additional, Fierlbeck, Gerhard, additional, Lakshminarayanan, Santhanam, additional, Allanore, Yannick, additional, Pope, Janet E, additional, Riemekasten, Gabriela, additional, Steen, Virginia, additional, Müller-Ladner, Ulf, additional, Spotswood, Helen, additional, Burke, Laura, additional, Siegel, Jeffrey, additional, Jahreis, Angelika, additional, and Furst, Daniel E, additional

Published
2017
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30. 306. A MULTICENTRE RELIABILITY AND VALIDITY STUDY OF LASER SPECKLE CONTRAST IMAGING AND THERMOGRAPHY IN PATIENTS WITH RAYNAUD’S PHENOMENON SECONDARY TO SYSTEMIC SCLEROSIS

Author

Murray, Andrea K., primary, Manning, Joanne, additional, Moore, Tonia, additional, Wilkinson, Jack, additional, Marjanovic, Elizabeth, additional, Leggett, Sarah, additional, Dinsdale, Graham, additional, Roberts, Christopher, additional, Allen, John, additional, Anderson, Marina E., additional, Britton, Jason, additional, Buch, Maya, additional, Del Galdo, Francesco, additional, Denton, Christopher P., additional, Drayton, Tracey, additional, Furlong, Anita, additional, Griffiths, Bridget, additional, Hall, Frances, additional, Hart, Darren, additional, Howell, Kevin, additional, MacDonald, Audrey, additional, McHugh, Neil J., additional, Pauling, John, additional, Shipley, Jacqueline, additional, and Herrick, Ariane, additional

Published
2017
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31. Treatment outcome in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study (ESOS)

Author

Herrick, Ariane L., Pan, Xiaoyan, Peytrignet, Sebastien, Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Brown, Edith, Czirjak, Laszlo, Distler, Joerg H. W., Distler, Oliver, Fligelstone, Kim, Gregory, William J., Ochiel, Rachel, Vonk, Madelon, Ancuta, Codrina, Ong, Voon H., Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Oyvind, Jordan, Alison C., Jobanputra, Paresh, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C., Agard, Christian, Anderson, Marina E., Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H., Chung, Lorinda, Damjanov, Nemanja, Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Soren, MacGregor, Alexander J., McHugh, Neil, Mueller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E., Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, Inanc, Murat, McLaren, John S., van Laar, Jacob M., Pathare, Sanjay, Proudman, Susannah, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J., Grange, Claire, Trad, Georges-Selim, Denton, Christopher P., Herrick, Ariane L., Pan, Xiaoyan, Peytrignet, Sebastien, Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Brown, Edith, Czirjak, Laszlo, Distler, Joerg H. W., Distler, Oliver, Fligelstone, Kim, Gregory, William J., Ochiel, Rachel, Vonk, Madelon, Ancuta, Codrina, Ong, Voon H., Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Oyvind, Jordan, Alison C., Jobanputra, Paresh, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C., Agard, Christian, Anderson, Marina E., Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H., Chung, Lorinda, Damjanov, Nemanja, Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Soren, MacGregor, Alexander J., McHugh, Neil, Mueller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E., Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, Inanc, Murat, McLaren, John S., van Laar, Jacob M., Pathare, Sanjay, Proudman, Susannah, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J., Grange, Claire, Trad, Georges-Selim, and Denton, Christopher P.

Abstract

Objectives The rarity of early diffuse cutaneous systemic sclerosis (dcSSc) makes randomised controlled trials very difficult. We aimed to use an observational approach to compare effectiveness of currently used treatment approaches. Methods This was a prospective, observational cohort study of early dcSSc (within three years of onset of skin thickening). Clinicians selected one of four protocols for each patient: methotrexate, mycophenolate mofetil (MMF), cyclophosphamide or 'no immunosuppressant'. Patients were assessed three-monthly for up to 24 months. The primary outcome was the change in modified Rodnan skin score (mRSS). Confounding by indication at baseline was accounted for using inverse probability of treatment (IPT) weights. As a secondary outcome, an IPT-weighted Cox model was used to test for differences in survival. Results Of 326 patients recruited from 50 centres, 65 were prescribed methotrexate, 118 MMF, 87 cyclophosphamide and 56 no immunosuppressant. 276 (84.7%) patients completed 12 and 234 (71.7%) 24 months follow-up (or reached last visit date). There were statistically significant reductions in mRSS at 12 months in all groups: -4.0 (-5.2 to -2.7) units for methotrexate, -4.1 (-5.3 to -2.9) for MMF, -3.3 (-4.9 to -1.7) for cyclophosphamide and -2.2 (-4.0 to -0.3) for no immunosuppressant (p value for between-group differences=0.346). There were no statistically significant differences in survival between protocols before (p=0.389) or after weighting (p=0.440), but survival was poorest in the no immunosuppressant group (84.0%) at 24 months. Conclusions These findings may support using immunosuppressants for early dcSSc but suggest that overall benefit is modest over 12 months and that better treatments are needed.

Published
2017

32. Treatment outcome in early diffuse cutaneous systemic sclerosis:the European Scleroderma Observational Study (ESOS)

Author

Herrick, Ariane L, Pan, Xiaoyan, Peytrignet, Sébastien, Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Brown, Edith, Czirják, László, Distler, Jörg H W, Distler, Oliver, Fligelstone, Kim, Gregory, William J, Ochiel, Rachel, Vonk, Madelon, Ancuţa, Codrina, Ong, Voon H, Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Øyvind, Jordan, Alison C, Jobanputra, Paresh, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C, Agard, Christian, Anderson, Marina E, Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H, Chung, Lorinda, Damjanov, Nemanja, Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Søren, MacGregor, Alexander J, McHugh, Neil, Müller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E, Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, Inanc, Murat, McLaren, John S, van Laar, Jacob M, Pathare, Sanjay, Proudman, Susannah, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J, Grange, Claire, Trad, Georges-Selim, Denton, Christopher P, Herrick, Ariane L, Pan, Xiaoyan, Peytrignet, Sébastien, Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Brown, Edith, Czirják, László, Distler, Jörg H W, Distler, Oliver, Fligelstone, Kim, Gregory, William J, Ochiel, Rachel, Vonk, Madelon, Ancuţa, Codrina, Ong, Voon H, Farge, Dominique, Hudson, Marie, Matucci-Cerinic, Marco, Balbir-Gurman, Alexandra, Midtvedt, Øyvind, Jordan, Alison C, Jobanputra, Paresh, Stevens, Wendy, Moinzadeh, Pia, Hall, Frances C, Agard, Christian, Anderson, Marina E, Diot, Elisabeth, Madhok, Rajan, Akil, Mohammed, Buch, Maya H, Chung, Lorinda, Damjanov, Nemanja, Gunawardena, Harsha, Lanyon, Peter, Ahmad, Yasmeen, Chakravarty, Kuntal, Jacobsen, Søren, MacGregor, Alexander J, McHugh, Neil, Müller-Ladner, Ulf, Riemekasten, Gabriela, Becker, Michael, Roddy, Janet, Carreira, Patricia E, Fauchais, Anne Laure, Hachulla, Eric, Hamilton, Jennifer, Inanc, Murat, McLaren, John S, van Laar, Jacob M, Pathare, Sanjay, Proudman, Susannah, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J, Grange, Claire, Trad, Georges-Selim, and Denton, Christopher P

Abstract

OBJECTIVES: The rarity of early diffuse cutaneous systemic sclerosis (dcSSc) makes randomised controlled trials very difficult. We aimed to use an observational approach to compare effectiveness of currently used treatment approaches.METHODS: This was a prospective, observational cohort study of early dcSSc (within three years of onset of skin thickening). Clinicians selected one of four protocols for each patient: methotrexate, mycophenolate mofetil (MMF), cyclophosphamide or 'no immunosuppressant'. Patients were assessed three-monthly for up to 24 months. The primary outcome was the change in modified Rodnan skin score (mRSS). Confounding by indication at baseline was accounted for using inverse probability of treatment (IPT) weights. As a secondary outcome, an IPT-weighted Cox model was used to test for differences in survival.RESULTS: Of 326 patients recruited from 50 centres, 65 were prescribed methotrexate, 118 MMF, 87 cyclophosphamide and 56 no immunosuppressant. 276 (84.7%) patients completed 12 and 234 (71.7%) 24 months follow-up (or reached last visit date). There were statistically significant reductions in mRSS at 12 months in all groups: -4.0 (-5.2 to -2.7) units for methotrexate, -4.1 (-5.3 to -2.9) for MMF, -3.3 (-4.9 to -1.7) for cyclophosphamide and -2.2 (-4.0 to -0.3) for no immunosuppressant (p value for between-group differences=0.346). There were no statistically significant differences in survival between protocols before (p=0.389) or after weighting (p=0.440), but survival was poorest in the no immunosuppressant group (84.0%) at 24 months.CONCLUSIONS: These findings may support using immunosuppressants for early dcSSc but suggest that overall benefit is modest over 12 months and that better treatments are needed.TRIAL REGISTRATION NUMBER: NCT02339441.

Published
2017

33. Treatment outcome in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study (ESOS)

Author

Herrick, Ariane L, primary, Pan, Xiaoyan, additional, Peytrignet, Sébastien, additional, Lunt, Mark, additional, Hesselstrand, Roger, additional, Mouthon, Luc, additional, Silman, Alan, additional, Brown, Edith, additional, Czirják, László, additional, Distler, Jörg H W, additional, Distler, Oliver, additional, Fligelstone, Kim, additional, Gregory, William J, additional, Ochiel, Rachel, additional, Vonk, Madelon, additional, Ancuţa, Codrina, additional, Ong, Voon H, additional, Farge, Dominique, additional, Hudson, Marie, additional, Matucci-Cerinic, Marco, additional, Balbir-Gurman, Alexandra, additional, Midtvedt, Øyvind, additional, Jordan, Alison C, additional, Jobanputra, Paresh, additional, Stevens, Wendy, additional, Moinzadeh, Pia, additional, Hall, Frances C, additional, Agard, Christian, additional, Anderson, Marina E, additional, Diot, Elisabeth, additional, Madhok, Rajan, additional, Akil, Mohammed, additional, Buch, Maya H, additional, Chung, Lorinda, additional, Damjanov, Nemanja, additional, Gunawardena, Harsha, additional, Lanyon, Peter, additional, Ahmad, Yasmeen, additional, Chakravarty, Kuntal, additional, Jacobsen, Søren, additional, MacGregor, Alexander J, additional, McHugh, Neil, additional, Müller-Ladner, Ulf, additional, Riemekasten, Gabriela, additional, Becker, Michael, additional, Roddy, Janet, additional, Carreira, Patricia E, additional, Fauchais, Anne Laure, additional, Hachulla, Eric, additional, Hamilton, Jennifer, additional, İnanç, Murat, additional, McLaren, John S, additional, van Laar, Jacob M, additional, Pathare, Sanjay, additional, Proudman, Susannah, additional, Rudin, Anna, additional, Sahhar, Joanne, additional, Coppere, Brigitte, additional, Serratrice, Christine, additional, Sheeran, Tom, additional, Veale, Douglas J, additional, Grange, Claire, additional, Trad, Georges-Selim, additional, and Denton, Christopher P, additional

Published
2017
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34. Safety and efficacy of subcutaneous tocilizumab in adults with systemic sclerosis (faSScinate): a phase 2, randomised, controlled trial

Author

MS Reumatologie/Immunologie/Infectie, Infection & Immunity, Khanna, Dinesh, Denton, Christopher P., Jahreis, Angelika, van Laar, Jacob M., Frech, Tracy M., Anderson, Marina E., Baron, Murray, Chung, Lorinda, Fierlbeck, Gerhard, Lakshminarayanan, Santhanam, Allanore, Yannick, Pope, Janet E., Riemekasten, Gabriela, Steen, Virginia, Müller-Ladner, Ulf, Lafyatis, Robert, Stifano, Giuseppina, Spotswood, Helen, Chen-Harris, Haiyin, Dziadek, Sebastian, Morimoto, Alyssa, Sornasse, Thierry, Siegel, Jeffrey, Furst, Daniel E., MS Reumatologie/Immunologie/Infectie, Infection & Immunity, Khanna, Dinesh, Denton, Christopher P., Jahreis, Angelika, van Laar, Jacob M., Frech, Tracy M., Anderson, Marina E., Baron, Murray, Chung, Lorinda, Fierlbeck, Gerhard, Lakshminarayanan, Santhanam, Allanore, Yannick, Pope, Janet E., Riemekasten, Gabriela, Steen, Virginia, Müller-Ladner, Ulf, Lafyatis, Robert, Stifano, Giuseppina, Spotswood, Helen, Chen-Harris, Haiyin, Dziadek, Sebastian, Morimoto, Alyssa, Sornasse, Thierry, Siegel, Jeffrey, and Furst, Daniel E.

Published
2016

35. Nailfold capillaroscopy—how many fingers should be examined to detect abnormality?

Author

Dinsdale, Graham, Roberts, Chris, Moore, Tonia, Manning, Joanne, Berks, Michael, Allen, John, Anderson, Marina E, Cutolo, Maurizio, Hesselstrand, Roger, Howell, Kevin, Pizzorni, Carmen, Smith, Vanessa, Sulli, Alberto, Wildt, Marie, Taylor, Christopher, Murray, Andrea, and Herrick, Ariane L

Subjects
RAYNAUD'S disease, SYSTEMIC scleroderma, ANGIOSCOPY, BIOMARKERS, FINGERS, DIAGNOSIS
Abstract

Objectives Nailfold capillaroscopy is being increasingly used by rheumatologists in the diagnosis of SSc. However, assessment of all nailfolds can be time-consuming in a busy outpatient clinic. Our aim was to answer the question as to how many (and which) fingers a clinician should routinely assess to capture accurately the true state. Methods A total of 2994 assessments (by an international panel of expert observers) of 1600 images from 173 participants (101 with SSc, 22 with primary RP and 50 healthy controls) were included in this analysis. Seven single-finger or finger combinations (derived from the middle and ring fingers) were then tested for sensitivity for the presence of two markers of capillary abnormality [presence of giant capillaries and an SSc grade (early, active or late)] compared with assessment of all eight fingers. Results For the eight-finger gold standard, sensitivity against the diagnostic criteria was 74.6% (53.0% for the presence of giants alone and 73.1% for image grade alone). Examining only one finger gave low sensitivity (ranging from right middle 31.7% to left ring 46.6%). Examining both ring fingers gave a sensitivity of 59.8%, whereas examining the four-finger combination of both ring and both middle fingers gave a sensitivity of 66.7%. Conclusion During routine capillaroscopic examination, ideally all eight nailbeds (excluding thumbs) should be examined, otherwise some abnormalities will be missed. Examining only four fingers reduces capillaroscopy sensitivity. [ABSTRACT FROM AUTHOR]

Published
2019
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37. Patient acceptable symptom state in scleroderma: results from the tocilizumab compared with placebo trial in active diffuse cutaneous systemic sclerosis.

Author

Arnold, Michael B, Khanna, Dinesh, Denton, Christopher P, Laar, Jacob M van, Frech, Tracy M, Anderson, Marina E, Baron, Murray, Chung, Lorinda, Fierlbeck, Gerhard, and Lakshminarayanan, Santhanam

Subjects
TOCILIZUMAB, BLOOD sedimentation, C-reactive protein, CLINICAL trials, HEALTH outcome assessment, QUESTIONNAIRES, SYSTEMIC scleroderma, VISUAL analog scale, ADULTS, THERAPEUTICS
Abstract

Objectives. Patient acceptable symptom state (PASS) as an absolute state of well-being has shown promise as an outcome measure in many rheumatologic conditions. We aimed to assess whether PASS may be effective in active diffuse cutaneous SSc differentiating active from placebo. Methods. Data from the phase 2 Safety and Efficacy of Subcutaneous Tocilizumab in Adults with Systemic Sclerosis (faSScinate) trial were used, which compared tocilizumab (TCZ) vs placebo over 48 weeks followed by an open-label TCZ period to 96 weeks. Three different types of PASS questions were evaluated at weeks 8, 24, 48 and 96, including if a current state would be acceptable over time as a yes vs no response and Likert scales about how acceptable a current state is if remaining over time. Additional outcomes assessed included modified Rodnan skin score, HAQ disability index (HAQ-DI), physician and patient global assessments on a visual analogue scale, CRP and ESR. Results. The placebo group consisted of 44 patients and the TCZ group had 43 patients. At baseline, 33% achieved a PASS for all three PASS questions, with the proportion increasing to 69, 71 and 78%, respectively, at 96 weeks. Changes in PASS scores showed a moderately negative correlation with HAQ-DI and patient and physician global assessments visual analogue scales, which indicates expected improvements as PASS improved. The PASS question, 'Considering all of the ways your scleroderma has affected you, how acceptable would you rate your level of symptoms?' showed significant correlations with patient-reported outcomes and differentiating placebo vs TCZ at 48 weeks (P = 0.023). Conclusion. PASS may be used as a patient-centred outcome in SSc, especially as a 7-point Likert scale. Further validation is required to determine the utility as an outcome measure in trials and clinical practice. [ABSTRACT FROM AUTHOR]

Published
2018
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40. Reconciling Healthcare Professional and Patient Perspectives in the Development of Disease Activity and Response Criteria in Connective Tissue Disease–related Interstitial Lung Diseases

Author

Saketkoo, Lesley Ann, Mittoo, Shikha, Frankel, Sid, LeSage, Daphne, Sarver, Catherine, Phillips, Kristine, Strand, Vibeke, Matteson, Eric L., Baughman, Robert P., Brown, Kevin K., Christmann, Romy B., Dellaripa, Paul, Denton, Christopher P., Distler, Oliver, Fischer, Aryeh, Flaherty, Kevin, Huscher, Dörte, Khanna, Dinesh, Kowal-Bielecka, Otylia, Merkel, Peter A., Oddis, Chester V., Pittrow, David, Sandorfi, Nora, Seibold, James R., Swigris, Jeffrey, Wells, Athol, Antoniou, Katerina, Castelino, Flavia V., Christopher-Stine, Lisa, Collard, Harold R., Cottin, Vincent, Danoff, Sonye, Hedlund, Robert, Highland, Kristin B., Hummers, Laura, Lynch, David A., Kim, Dong Soon, Ryu, Jay H., Christopher-Stine, Lisa, Miller, Frederick W., Nichols, Karen, Proudman, Susanna M., Richeldi, Luca, Shah, Ami A., van den Assum, Pieter, Aggarwal, Rohit, Ainslie, Gillian, Alkassab, Firas, Allanore, Yannick, Anderson, Marina E., Andonopoulos, Andrew P., Antin-Ozerkis, Danielle, Antoniou, Katerina, Arrobas, Ana, Ascherman, Dana P., Assassi, Shervin, Baron, Murray, Bathon, Joan M., Baughman, Robert P., Behr, Juergen, Beretta, Lorenzo, Bingham, Clifton O., Binnie, Matthew, Birring, Surinder S., Boin, Francesco, Bongartz, Tim, Bourdin, Arnaud, Bouros, Demosthenes, Brasington, Richard, Bresser, Paul, Brown, Kevin K., Buch, Maya H., Burge, P. Sherwood, Carmona, Loreto, Castelino, Flavia V., Carreira, Patricia E., Carvalho, Carlos R.R., Catoggio, Luis J., Chan, Kevin M., Chapman, Jeffrey, Chatterjee, Soumya, Christmann, Romy B., Christopher-Stine, Lisa, Chua, Felix, Chung, Lorinda, Conron, Matthew, Corte, Tamera, Cosgrove, Gregory, Costabel, Ulrich, Cottin, Vincent, Cox, Gerard, Crestani, Bruno, Crofford, Leslie J., Csuka, Mary E., Curbelo, Pablo, Czirják, László, Daniil, Zoe, Danoff, Sonye, D’Arsigny, Christine L., Davis, Gerald S., de Andrade, Joao A., Dellaripa, Paul F., De Vuyst, Paul, Dempsey, Owen J., Denton, Christopher P., Derk, Chris T., Distler, Jörg, Distler, Oliver, Dixon, William G., Downey, Gregory, Doyle, Mittie K., Drent, Marjolein, Durairaj, Lakshmi, Emery, Paul, Espinoza, Luis R., Farge, Dominique, Fathi, Maryam, Fell, Charlene D., Fessler, Barri J., Fischer, Aryeh, Fitzgerald, John E., Flaherty, Kevin R., Foeldvari, Ivan, Fox, George A., Frech, Tracy M., Freitas, Sara, Furst, Daniel E., Gabrielli, Armando, García-Vicuña, Rosario, Georgiev, Ognian B., Gerbino, Anthony, Gillisen, Adrian, Gladman, Dafna D., Glassberg, Marilyn, Gochuico, Bernadette R., Gogali, Athena, Goh, Nicole S., Goldberg, Avram, Goldberg, Hilary J., Gourley, Mark F., Griffing, Leroy, Grutters, Jan C., Gunnarsson, Ragnar, Hachulla, Eric, Hall, Frances C., Harari, Sergio, Herrick, Ariane L., Herzog, Erica L., Hesselstrand, Roger, Highland, Kristin, Hirani, Nikhil, Hodgson, Ulla, Hollingsworth, Helen M., Homer, Robert J., Hoyles, Rachel K., Hsu, Vivien M., Hubbard, Richard B., Hummers, Laura, Hunzelmann, Nicolas, Huscher, Dörte, Isasi, Maria Eloisa, Isasi, Elida Susana, Jacobsen, Soren, Jimenez, Sergio A., Johnson, Sindhu R., Jones, Christine H., Kahaleh, Bashar, Kairalla, Ronaldo A., Kalluri, Meena, Kalra, Sanjay, Kaner, Robert J., Khanna, Dinesh, Kim, Dong Soon, Kinder, Brent W., Kiter, Goksel, Klingsberg, Ross C., Kokosi, Maria, Kolb, Martin R.J., Kowal-Bielecka, Otylia M., Kur-Zalewska, Joanna, Kuwana, Masataka, Lake, Fiona R., Lally, Edward V., Lasky, Joseph A., Laurindo, Ileda M., Able, Lawrence, Lee, Peter, Leonard, Colm T., Lien, Dale C., Limper, Andrew H., Liossis, Stamatis-Nick C., Lohr, Kristine M., Loyd, James E., Lundberg, Ingrid E., Mageto, Yolanda N., Maher, Toby M., Mahmud, Tafazzul H., Manganas, Helene, Marie, Isabelle, Marras, Theodore K., Martinez, José Antônio Baddini, Martinez, Fernando J., Mathieu, Alessandro, Matucci-Cerinic, Marco, Mayes, Maureen D., McKown, Kevin M., Medsger, Thomas A., Meehan, Richard T., Mendes, Ana Cristina, Merkel, Peter A., Meyer, Keith C., Millar, Ann B., Miller, Frederick W., Moğulkoç, Nesrin, Molitor, Jerry A., Morais, António, Mouthon, Luc, Müller, Veronika, Müller-Quernheim, Joachim, Nadashkevich, Oleg, Nador, Roland, Nash, Peter, Nathan, Steven D., Navarro, Carmen, Neves, Sofia, Noth, Imre, Nunes, Hilario, Oddis, Chester V., Olson, Amy L., Opitz, Christian F., Padilla, Maria, Pappas, Dimitrios, Parfrey, Helen, Pego-Reigosa, José M., Pereira, Carlos A.C., Perez, Rafael, Phillips, Kristine, Pittrow, David, Pope, Janet E., Porter, Joanna C., Proudman, Susanna M., Renzoni, Elisabeth A., Richeldi, Luca, Riemekasten, Gabriela, Riley, David J., Rischmueller, Maureen, Rodriguez-Reyna, Tatiana S., Rojas-Serrano, J., Roman, Jesse, Rosen, Glenn D., Rossman, Milton, Rothfield, Naomi, Ryu, Jay H., Sahn, Steven A., Sandorfi, Nora, Sanduzzi, Alessandro, Scholand, Mary Beth, Seibold, James R., Selman, Moises, Senécal, Jean-Luc, Seo, Philip, Shah, Ami, Silver, Richard M., Solomon, Joshua J., Steen, Virginia, Stevens, Wendy, Strange, Charlie, Sussman, Robert, Sutton, Evelyn D., Sweiss, Nadera J., Swigris, Jeffrey, Tornling, Göran, Tzelepis, George E., Undurraga, Alvaro, Vacca, Allessandra, Vancheri, Carlo, Varga, Janos, Veale, Douglas J., Volkov, Suncica, Walker, Ulrich A., Wells, Athol U., Wencel, Mark, Wesselius, Lewis J., Wickremasinghe, Melissa, Wilcox, Pearce, Wilsher, Margaret L., Wollheim, Frank A., Wuyts, Wim A., Yung, Gordon, Zanon, Pietro, Zappala, Christopher J., Groshong, Steve D., Leslie, Kevin O., Myers, Jeffrey L., Padera, Robert F., Desai, Sujal R., Goldin, Jonathan, Kazerooni, Ella A., Klein, Jeffrey S., Lynch, David A., and Keen, Kevin J.

Abstract

Interstitial lung diseases (ILD), including those related to connective tissue disease (CTD), and idiopathic pulmonary fibrosis (IPF) carry high morbidity and mortality. Great efforts are under way to develop and investigate meaningful treatments in the context of clinical trials. However, efforts have been challenged by a lack of validated outcome measures and by inconsistent use of measures in clinical trials. Lack of consensus has fragmented effective use of strategies in CTD-ILD and IPF, with a history of resultant difficulties in obtaining agency approval of treatment interventions. Until recently, the patient perspective to determine domains and outcome measures in CTD-ILD and IPF had never been applied. Efforts described here demonstrate unequivocally the value and influence of patient involvement on core set development. Regarding CTD-ILD, this is the first OMERACT working group to directly address a manifestation/comorbidity of a rheumatic disease (ILD) as well as a disease not considered rheumatic (IPF). The OMERACT 11 proceedings of the CTD-ILD Working Group describe the forward and lateral process to include both the medical and patient perspectives in the urgently needed identification of a core set of preliminary domains and outcome measures in CTD-ILD and IPF.

Published
2014
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41. Defining the neutrophil phenotype in systemic sclerosis

Author

Barnes, Theresa, Moots, Robert J., Edwards, Steven W., and Anderson, Marina E.

Subjects
610, R Medicine (General)
Abstract

Background: Systemic sclerosis (SSc) is a disease characterised by a triad of immunological abnormalities, endothelial cell dysfunction and fibrosis. Neutrophils are the most abundant circulating leukocyte. They contain several mediators which when released can lead to modulation of the inflammatory response, cause endothelial cell activation and injury and eventually lead to fibrosis. The peri-endothelial cell environment in SSc has the potential to lead to neutrophil activation and indeed this has been previously described in the literature in terms of reactive oxygen species (ROS) generation. In this thesis I aim to explore the hypothesis that: “Neutrophils are activated in SSc and contribute to endothelial cell activation and damage”. Methods: The functional phenotype of SSc neutrophils was explored in vitro. Functions investigated included; ROS generation, chemotaxis, integrin expression, apoptosis and CD16 expression. The protein expression of ex vivo SSc neutrophils was compared to healthy control neutrophils. The DIGE technology was used to explore the pan-proteome and iTRAQ was used to focus in on the plasma membrane proteome. The role of neutrophil elastase was explored in SSc by examining the serum neutrophil elastase concentration and activity and correlating these to clinical manifestations of disease. The role of neutrophil: endothelial cell interactions was modelled in vitro using live cell imaging by confocal microscopy looking for evidence of endothelial cell activation (E-selectin expression) and apoptosis. Experiments examined the role of neutrophil derived mediators in these interactions. Results: Functional studies revealed that SSc neutrophils are hypofunctional in terms of spontaneous ROS generation and chemotaxis in vitro. This may reflect in vivo activation. SSc neutrophils are similar in terms of integrin expression and baseline apoptosis to control neutrophils. Pan-proteomic studies reveal neutrophil activation in SSc since changes in protein expression mirror those seen in response to neutrophil activators TNFα and LPS. Proteomic studies also point to neutrophil priming in vivo. Serum neutrophil elastase concentrations and activity were not elevated in SSc, however, discrepancies between concentration and activity suggest a functional deficiency in elastase inhibitors in SSc serum. Serum elastase activity and concentrations were found to be lower in RNP (ribonucleoprotein) positive patients indicating that different mechanisms maybe involved in different SSc subtypes. In vitro models demonstrate that SSc serum causes endothelial cell activation and apoptosis and that neutrophils are essential for this effect. Serine proteases seem to play an important role in inducing apoptosis and IL-6 trans signalling is involved in endothelial cell activation and apoptosis. Neutrophils do not express IL-6 but are dominant sources of the soluble IL-6R which is essential for trans signalling. Conclusions: There is evidence that neutrophils are activated in SSc though; no specific activating signature is identified. In co-cultures, neutrophils are essential for endothelial cell activation and apoptosis in response to SSc serum. Neutrophil mediators including serine proteases and IL6R are likely to play important roles in this effect. Therefore neutrophils may play an important part in the propagation of inflammation and endothelial cell activation which eventually leads to the fibrotic phenotype which is characteristic of this disease.

Published
2010
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42. Disability, fatigue, pain and their associates in early diffuse cutaneous systemic sclerosis: the European Scleroderma Observational Study.

Author

Peytrignet S, Denton CP, Lunt M, Hesselstrand R, Mouthon L, Silman A, Pan X, Brown E, Czirják L, Distler JHW, Distler O, Fligelstone K, Gregory WJ, Ochiel R, Vonk M, Ancuta C, Ong VH, Farge D, Hudson M, Matucci-Cerinic M, Balbir-Gurman A, Midtvedt Ø, Jordan AC, Stevens W, Moinzadeh P, Hall FC, Agard C, Anderson ME, Diot E, Madhok R, Akil M, Buch MH, Chung L, Damjanov N, Gunawardena H, Lanyon P, Ahmad Y, Chakravarty K, Jacobsen S, MacGregor AJ, McHugh N, Müller-Ladner U, Riemekasten G, Becker M, Roddy J, Carreira PE, Fauchais AL, Hachulla E, Hamilton J, Inanç M, McLaren JS, van Laar JM, Pathare S, Proudman S, Rudin A, Sahhar J, Coppere B, Serratrice C, Sheeran T, Veale DJ, Grange C, Trad GS, and Herrick AL

Subjects
Adult, Cost of Illness, Europe, Fatigue etiology, Female, Fingers, Hand Strength, Health Surveys, Humans, Male, Pain etiology, Prospective Studies, Scleroderma, Diffuse complications, Skin Ulcer etiology, Skin Ulcer physiopathology, Disability Evaluation, Fatigue physiopathology, Pain physiopathology, Scleroderma, Diffuse physiopathology, Severity of Illness Index
Abstract

Objectives: Our aim was to describe the burden of early dcSSc in terms of disability, fatigue and pain in the European Scleroderma Observational Study cohort, and to explore associated clinical features., Methods: Patients completed questionnaires at study entry, 12 and 24 months, including the HAQ disability index (HAQ-DI), the Cochin Hand Function Scale (CHFS), the Functional Assessment of Chronic Illness Therapy-fatigue and the Short Form 36 (SF36). Associates examined included the modified Rodnan skin score (mRSS), current digital ulcers and internal organ involvement. Correlations between 12-month changes were also examined., Results: The 326 patients recruited (median disease duration 11.9 months) displayed high levels of disability [mean (s.d.) HAQ-DI 1.1 (0.83)], with 'grip' and 'activity' being most affected. Of the 18 activities assessed in the CHFS, those involving fine finger movements were most affected. High HAQ-DI and CHFS scores were both associated with high mRSS (ρ = 0.34, P < 0.0001 and ρ = 0.35, P < 0.0001, respectively). HAQ-DI was higher in patients with digital ulcers (P = 0.004), pulmonary fibrosis (P = 0.005), cardiac (P = 0.005) and muscle involvement (P = 0.002). As anticipated, HAQ-DI, CHFS, the Functional Assessment of Chronic Illness Therapy and SF36 scores were all highly correlated, in particular the HAQ-DI with the CHFS (ρ = 0.84, P < 0.0001). Worsening HAQ-DI over 12 months was strongly associated with increasing mRSS (ρ = 0.40, P < 0.0001), decreasing hand function (ρ = 0.57, P < 0.0001) and increasing fatigue (ρ = -0.53, P < 0.0001)., Conclusion: The European Scleroderma Observational Study highlights the burden of disability in early dcSSc, with high levels of disability and fatigue, associating with the degree of skin thickening (mRSS). Impaired hand function is a major contributor to overall disability., (© The Author 2017. Published by Oxford University Press on behalf of the British Society for Rheumatology. All rights reserved. For Permissions, please email: journals.permissions@oup.com)

Published
2018
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43. Computerized nailfold video capillaroscopy--a new tool for assessment of Raynaud's phenomenon.

Author

Anderson ME, Allen PD, Moore T, Hillier V, Taylor CJ, and Herrick AL

Subjects
Adult, Aged, Aged, 80 and over, Capillaries physiology, Female, Humans, Male, Microscopic Angioscopy statistics & numerical data, Microscopy, Video, Middle Aged, Nails blood supply, Observer Variation, Raynaud Disease physiopathology, Scleroderma, Systemic diagnosis, Scleroderma, Systemic physiopathology, Software, Image Processing, Computer-Assisted methods, Microscopic Angioscopy instrumentation, Microscopic Angioscopy methods, Raynaud Disease diagnosis
Abstract

Objective: To develop a computer based nailfold video capillaroscopy system with enhanced image quality and to assess its disease-subgroup resolving power in patients with primary and secondary Raynaud's phenomenon (RP)., Methods: Using frame registration software, digitized video images from the microscope were combined to form a panoramic mosaic of the nailfold. Capillary dimensions (apex, arterial, venous, and total width) and density were measured onscreen. Significantly, the new system could guarantee analysis of the same set of capillaries by 2 observers. Forty-eight healthy control subjects, 21 patients with primary RP, 40 patients with limited cutaneous systemic sclerosis (lcSSc), and 11 patients with diffuse cutaneous SSc (dcSSc) were studied. Intra- and interobserver variability were calculated in a subset of 30 subjects., Results: The number of loops/mm was significantly lower, and all 4 capillary dimensions significantly greater, in SSc patients versus controls plus primary RP patients (p < 0.001 for all measures). When comparing control (+ primary RP) patients with SSc patients (lcSSc + dcSSc) the most powerful discriminator was found to be the number of loops/mm. Results for intra- and interobserver reproducibility showed that the limits of agreement were closer when both observers measured the same capillaries., Conclusion: The key feature of the newly developed system is that it improves reproducibility of nailfold capillary measurements by allowing reidentification of the same capillaries by different observers. By allowing access to previous measurements, the new system should improve reliability in longitudinal studies, and therefore has the potential of being a valuable outcome measure of microvessel disease/involvement in clinical trials of scleroderma spectrum disorders.

Published
2005

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