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2. The scale-dependent role of submerged macrophytes as drift-feeding lotic fish habitat

Author

McLaren, John S., Van Kirk, Robert W., Budy, Phaedra, and Brothers, Soren

Subjects
Aquatic plants -- Environmental aspects, Rainbow trout -- Environmental aspects, Rivers -- Environmental aspects, Earth sciences
Abstract

Although submerged macrophyte (hereafter, 'macrophyte') communities are globally prevalent in low-gradient rivers, the net reach-scale effect of macrophytes on drift-feeding fish microhabitat preference is poorly understood. We used snorkeling and bioenergetics to study fish habitat selection for rainbow trout (Oncorhynchus mykiss) in the Henrys Fork, ID, USA, investigating microhabitat preference across a reach-scale gradient of macrophyte growth. Fish preferred microhabitats with deep water, low velocity, and low macrophyte coverage. Preferences for microhabitats with higher net rate of energy intake (NREI) were modulated by reach-scale macrophyte coverage, higher coverage increasing preferences for higher NREI. Macrophyte coverage was a weak positive predictor for depth and NREI, and a weak negative predictor for water velocity and median substrate. Our results suggest trade-offs between fish predation risk and bioenergetic food intake, with macrophytes modulating these trade-offs across scales by affecting reach-scale geomorphology, bioenergetics, and predation risk. As such, this study highlights the important and dynamic role that macrophytes can play in fish population dynamics in rivers, with important implications for management decisions. Key words: stream ecology, fish habitat, space use ecology, submerged macrophytes, bioenergetics, microhabitats, Introduction Clear, low-gradient lotic ecosystems throughout the world offer suitable conditions for the proliferation of submerged macrophytes, often reaching >50% areal coverage of the channel (Haslam 1978; Canfield and Hoyer [...]

Published
2023
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3. Drawdown, Habitat, and Kokanee Populations in a Western U.S. Reservoir.

Author

McLaren, John S., Van Kirk, Robert W., Mabaka, Arthur J., Brothers, Soren, and Budy, Phaedra

Subjects
RESERVOIR drawdown, SOCKEYE salmon, FISH populations, FISH habitats, HABITATS
Abstract

Greater drought frequency and severity due to climate change will result in greater drawdown of water storage reservoirs. However, changes to oxythermal regimes due to drawdown are reservoir specific and interface with fish species‐specific habitat requirements, producing varying effects on coldwater fish populations. We examined the effect of drawdown on the oxythermal habitat and relative abundance of kokanee Oncorhynchus nerka, a coldwater salmonid, in Island Park Reservoir on the Henrys Fork of the Snake River, Idaho. A measure of relative kokanee abundance was negatively, exponentially related to drawdown. Oxythermal patterns measured in the reservoir during 2021, a severe drought year, revealed that drawdown reduced kokanee habitat by increasing water temperatures and decreasing dissolved oxygen concentrations. Oxythermal refugia for kokanee appeared to relate to inflow from the spring‐fed Henrys Fork and other groundwater inflows. However, we did not quantify groundwater flow or connections, and we did not study kokanee population demographics or mortality. Reducing these sources of uncertainty is a priority for future study. Still, our study highlights a potential mechanism connecting reservoir drawdown to fish populations and the unique yet predictable mechanisms by which reservoir drawdown interacts with reservoir morphometry to affect fish habitat availability. [ABSTRACT FROM AUTHOR]

Published
2023
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7. 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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8. 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

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

Author

Peytrignet, Sebastien, Denton, Christopher P., Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Pan, Xiaoyan, Brown, Edith, Czirjak, Laszlo, Distler, Jorg 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., 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, Muller-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, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J., Grange, Claire, Trad, Georges-Selim, Herrick, Ariane L., Peytrignet, Sebastien, Denton, Christopher P., Lunt, Mark, Hesselstrand, Roger, Mouthon, Luc, Silman, Alan, Pan, Xiaoyan, Brown, Edith, Czirjak, Laszlo, Distler, Jorg 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., 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, Muller-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, Rudin, Anna, Sahhar, Joanne, Coppere, Brigitte, Serratrice, Christine, Sheeran, Tom, Veale, Douglas J., Grange, Claire, Trad, Georges-Selim, and Herrick, Ariane L.

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 (rho = 0.34, P < 0.0001 and rho = 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 (rho = 0.84, P < 0.0001). Worsening HAQ-DI over 12 months was strongly associated with increasing mRSS (rho = 0.40, P < 0.0001), decreasing hand function (rho = 0.57, P < 0.0001) and increasing fatigue (rho = -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.

Published
2018

10. 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

11. 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

12. 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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13. 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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14. 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

15. 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

16. 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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17. 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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