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11. A Review of Sensor Node in Wireless Sensor Networks

Author

Jobanputra Paresh Ashokkumar and Arun Jhapate

Subjects
Energy efficiency, Bandwidth, Computer science, business.industry, Sensor node, Computer Engineering, Wireless Sensor Networks, business, Wireless sensor network, Computer network
Abstract

Wireless Sensor Networks WSNs are collection of tiny sensor nodes capable of sensing, processing and broadcasting data correlated to some occurrence in the network area. The sensor nodes have severe limitation, such as bandwidth, short communication range, limited CPU processing facility, memory and energy. Enhancing the lifetime of wireless sensors network and efficient utilizations of bandwidth are essential for the proliferation of wireless sensor network in different applications. We provide an in depth study of applying wireless sensor networks WSNs to real world habitat monitoring. A set of system design requirements were developed that cover the hardware design of the nodes, the sensor network software, protective enclosures, and system architecture to meet the requirements of biologists. Although researchers anticipate some challenges arising in real world deployments of WSNs, many problems can only be discovered through experience. We present a set of experiences from a four month long deployment on a remote island. We analyze the environmental and node health data to evaluate system performance. The close integration of WSNs with their environment provides environmental data at densities previously impossible. We show that the sensor data is also useful for predicting system operation and network failures. Based on over one million data readings, we analyze the node and network design and develop network reliability profiles and failure models. Jobanputra Paresh Ashokkumar | Prof. Arun Jhapate "A Review of Sensor Node in Wireless Sensor Networks" Published in International Journal of Trend in Scientific Research and Development (ijtsrd), ISSN: 2456-6470, Volume-3 | Issue-4 , June 2019, URL: https://www.ijtsrd.com/papers/ijtsrd23620.pdf

Published
2019

14. Patterns of analgesic use, pain and self-efficacy: a cross-sectional study of patients attending a hospital rheumatology clinic

Author

Greenfield Sheila, Jolly Kate, Blamey Ruth, and Jobanputra Paresh

Subjects
Diseases of the musculoskeletal system, RC925-935
Abstract

Abstract Background Many people attending rheumatology clinics use analgesics and non-steroidal anti-inflammatories for persistent musculoskeletal pain. Guidelines for pain management recommend regular and pre-emptive use of analgesics to reduce the impact of pain. Clinical experience indicates that analgesics are often not used in this way. Studies exploring use of analgesics in arthritis have historically measured adherence to such medication. Here we examine patterns of analgesic use and their relationships to pain, self-efficacy and demographic factors. Methods Consecutive patients were approached in a hospital rheumatology out-patient clinic. Pattern of analgesic use was assessed by response to statements such as 'I always take my tablets every day.' Pain and self-efficacy (SE) were measured using the Western Ontario and McMaster Universities Osteoarthritis Index (WOMAC) and Arthritis Self-Efficacy Scale (ASES). Influence of factors on pain level and regularity of analgesic use were investigated using linear regression. Differences in pain between those agreeing and disagreeing with statements regarding analgesic use were assessed using t-tests. Results 218 patients (85% of attendees) completed the study. Six (2.8%) patients reported no current pain, 26 (12.3%) slight, 100 (47.4%) moderate, 62 (29.4%) severe and 17 (8.1%) extreme pain. In multiple linear regression self efficacy and regularity of analgesic use were significant (p < 0.01) with lower self efficacy and more regular use of analgesics associated with more pain. Low SE was associated with greater pain: 40 (41.7%) people with low SE reported severe pain versus 22 (18.3%) people with high SE, p < 0.001. Patients in greater pain were significantly more likely to take analgesics regularly; 13 (77%) of those in extreme pain reported always taking their analgesics every day, versus 9 (35%) in slight pain. Many patients, including 46% of those in severe pain, adjusted analgesic use to current pain level. In simple linear regression, pain was the only variable significantly associated with regularity of analgesic use: higher levels of pain corresponded to more regular analgesic use (p = 0.003). Conclusion Our study confirms that there is a strong inverse relationship between self-efficacy and pain severity. Analgesics are often used irregularly by people with arthritis, including some reporting severe pain.

Published
2009
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15. Disseminated cutaneous Herpes Simplex Virus-1 in a woman with rheumatoid arthritis receiving Infliximab: A case report

Author

Justice Elizabeth, Khan Sophia, Logan Sarah, and Jobanputra Paresh

Subjects
Medicine
Abstract

Abstract Introduction We present the case of a 49-year-old woman with a seronegative rheumatoid arthritis who developed pustular psoriasis whilst on etanercept and subsequently developed disseminated herpes simplex on infliximab. Case presentation Our patient presented with an inflammatory arthritis which failed to respond to both methotrexate and leflunomide, and sulphasalazine treatment led to side effects. She was started on etanercept but after 8 months of treatment developed scaly pustular lesions on her palms and soles typical of pustular psoriasis. Following the discontinuation of etanercept, our patient required high doses of oral prednisolone to control her inflammatory arthritis. A second biologic agent, infliximab, was introduced in addition to low-dose methotrexate and 15 mg of oral prednisolone. However, after just 3 infusions of infliximab, she was admitted to hospital with a fever, widespread itchy vesicular rash and worsening inflammatory arthritis. Fluid from skin vesicles examined by polymerase chain reaction showed Herpes Simplex Virus type 1. Blood cultures were negative and her chest X-ray was normal. Her infliximab was discontinued and she was started on acyclovir, 800 mg five times daily for 2 weeks. She made a good recovery with improvement in her skin within 48 hours. She continued for 2 months on a prophylactic dose of 400 mg bd. Her rheumatoid arthritis became increasingly active and a decision was made to introduce adalimumab alongside acyclovir. Acyclovir prophylaxis has been continued but the dose tapered so that she is taking only 200 mg of acyclovir on alternate days. There has been no recurrence of Herpes Simplex Virus lesions despite increasing adalimumab to 40 mg weekly 3 months after starting treatment. Conclusion We believe this to be the first reported case of widespread cutaneous Herpes Simplex Virus type 1 infection following treatment with infliximab. We discuss the clinical manifestations of Herpes Simplex Virus infections with particular emphasis on the immunosuppressed patient and the use of prophylactic acyclovir. Pustular psoriasis is now a well recognised but uncommon side effect of antitumour necrosis factor therapy and can lead to cessation of therapy, as in our patient's case.

Published
2008
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17. Hepatotoxicity associated with sulfasalazine in inflammatory arthritis: A case series from a local surveillance of serious adverse events

Author

Rankin Elizabeth, Bowman Simon, Homer Dawn, Maggs Fiona, Amarasena Roshan, Jobanputra Paresh, Filer Andrew, Raza Karim, and Jubb Ronald

Subjects
Diseases of the musculoskeletal system, RC925-935
Abstract

Abstract Background Spontaneous reporting systems for adverse drug reactions (ADRs) are handicapped by under-reporting and limited detail on individual cases. We report an investigation from a local surveillance for serious adverse drug reactions associated with disease modifying anti-rheumatic drugs that was triggered by the occurrence of liver failure in two of our patients. Methods Serious ADR reports have been solicited from local clinicians by regular postcards over the past seven years. Patients', who had hepatotoxicity on sulfasalazine and met a definition of a serious ADR, were identified. Two clinicians reviewed structured case reports and assessed causality by consensus and by using a causality assessment instrument. The likely frequency of hepatotoxicity with sulfasalazine was estimated by making a series of conservative assumptions. Results Ten cases were identified: eight occurred during surveillance. Eight patients were hospitalised, two in hepatic failure – one died after a liver transplant. All but one event occurred within 6 weeks of treatment. Seven patients had a skin rash, three eosinophilia and one interstitial nephritis. Five patients were of Black British of African or Caribbean descent. Liver enzymes showed a hepatocellular pattern in four cases and a mixed pattern in six. Drug-related hepatotoxicity was judged probable or highly probable in 8 patients. The likely frequency of serious hepatotoxicity with sulfasalazine was estimated at 0.4% of treated patients. Conclusion Serious hepatotoxicity associated with sulfasalazine appears to be under-appreciated and intensive monitoring and vigilance in the first 6 weeks of treatment is especially important.

Published
2008
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19. A pragmatic randomised controlled trial of hydrotherapy and land exercises on overall well being and quality of life in rheumatoid arthritis

Author

Nightingale Peter, Maggs Fiona, Eversden Lis, and Jobanputra Paresh

Subjects
Diseases of the musculoskeletal system, RC925-935
Abstract

Abstract Background Hydrotherapy is highly valued by people with rheumatoid arthritis yet few studies have compared the benefits of exercises in heated water against exercises on land. In particular, data on quality of life is rarely reported. This is especially important because patients treated with hydrotherapy often report an enhanced sense of well-being. We report a randomised controlled trial in which we compared the effects of hydrotherapy with exercises on land on overall response to treatment, physical function and quality of life in patients with rheumatoid arthritis. Methods One hundred and fifteen patients with RA were randomised to receive a weekly 30-minute session of hydrotherapy or similar exercises on land for 6 weeks. Our primary outcome was a self-rated global impression of change – a measure of treatment effect on a 7-point scale ranging from 1(very much worse) to 7 (very much better) assessed immediately on completion of treatment. Secondary outcomes including EuroQol health related quality of life, EuroQol health status valuation, HAQ, 10 metre walk time and pain scores were collected at baseline, after treatment and 3 months later. Binary outcomes were analysed by Fisher's exact test and continuous variables by Wilcoxon or Mann-Whitney tests. Results Baseline characteristics of the two groups were comparable. Significantly more patients treated with hydrotherapy (40/46, 87%) were much better or very much better than the patients treated with land exercise (19/40, 47.5%), p < 0.001 Fisher's exact test. Eleven patients allocated land exercise failed to complete treatment compared with 4 patients allocated hydrotherapy (p = 0.09). Sensitivity analyses confirmed an advantage for hydrotherapy if we assumed non-completers would all not have responded (response rates 70% versus 38%; p < 0.001) or if we assumed that non-completers would have had the same response as completers (response rates 82% versus 55% p = 0.002). Ten metre walk time improved after treatment in both cases (median pre-treatment time for both groups combined 10.9 seconds, post-treatment 9.1 s, and 3 months later 9.6 s). There was however no difference between treatment groups. Similarly there were no significant differences between groups in terms of changes to HAQ, EQ-5D utility score, EQ VAS and pain VAS. Conclusion Patients with RA treated with hydrotherapy are more likely to report feeling much better or very much better than those treated with land exercises immediately on completion of the treatment programme. This perceived benefit was not reflected by differences between groups in 10-metre walk times, functional scores, quality of life measures and pain scores.

Published
2007
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20. 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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21. BHPR research: qualitative1. Complex reasoning determines patients' perception of outcome following foot surgery in rheumatoid arhtritis

Author

Backhouse, Michael R., Vinall, Karen A., Redmond, Anthony, Helliwell, Philip, Keenan, Anne-Maree, Dale, Rebecca M., Thomas, Amanda, Aronson, Diane, Turner-Cobb, Julie, Sengupta, Raj, France, Brisa, Hill, Ingrid, Flurey, Caroline A., Morris, Marianne, Pollock, Jon, Hughes, Rod, Richards, Pam, Hewlett, Sarah, Ryan, Sarah, Lille, Kate, Adams, Jo, Haq, Inam, McArthur, Margaret, Goodacre, Lynne, Birt, Linda, Wilson, Oonagh, Kirwan, John, Dures, Emma, Quest, Enid, Rajak, Rizwan, Thomas, Tasmin, Lawson, Tom, Petford, Sharon, Hale, Elizabeth, Kitas, George D., Gooberman-Hill, Rachael, Jinks, Clare, Dziedzic, Krysia, Bouças, Sofia Barbosa, Hislop, Kelly, Rhodes, Carol, Ali, Fizzah, Ong, Bie Nio, White, Derrick, Hensor, Elizabeth, Ferguson, Anna M., Douiri, Abdel, Scott, David L., Lempp, Heidi, Halls, Serena, Law, Rebecca-Jane, Jones, Jeremy, Markland, David, Maddison, Peter, Thom, Jeanette, Thom, Jeanette M., Breslin, Anne, Kraus, Alexandra, Gordhan, Chandrika, Dennis, Sean, Connor, John, Chowdhary, Bhavani, Lottay, Neena, Juneja, Parmjit, Bacon, Paul A., Isaacs, David, Jack, James, Keller, Majella, Tibble, Jeremy, Hammond, Alison, Gill, Rachel, Tyson, Sarah, Tennant, Alan, Nordenskiold, Ulla, Pease, Emily E., Pease, Colin T., Trehane, Anne, Rahmeh, Fouz, Cornell, Patricia, Westlake, Sarah L., Rose, Katy, Alber, Catherine Fouad, Watson, Liam, Stratton, Richard, Lazarus, Mark, McNeilly, Natasha E., Waterfield, Jackie, Hurley, Mike, Greenwood, James, Clayton, Ann M., Lynch, Michael, Clewes, Adrian, Dawson, Julie, Abernethy, Veronica, Griffiths, Ann E., Chamberlain, Victoria A., McLoughlin, YokeMei, Campbell, Sophie, Hayes, Janita, Moffat, Claire, McKenna, Frank, Shah, Preeti, Williams, Ann, Rhys-Dillon, Ceril, Goodfellow, Rhian, Martin, James C., Bari, Farhan, Hughes, Gwenan, Thomas, Eleri, Baker, Sarah, Collins, David, Price, Elizabeth, Williamson, Lyn, Dunkley, Lisa, Youll, Matthew J., Rodziewicz, Mia, Reynolds, John A., Berry, Jacqueline, Pavey, Chee, Hyrich, Kimme, Gorodkin, Rachel, Wilkinson, Kate, Bruce, Ian, Barton, Anne, Silman, Alan, Ho, Pauline, Cornell, Tricia, Richards, Selwyn, Holmes, A., Parker, S., Smith, H., Briggs, N., Arthanari, S., Nisar, M., Thwaites, Catharine, Kamath, Sanjeet, Price, Shyra, Robinson, Sandra M., Walker, David, Coop, Hazel, Al-Allaf, Wahab, Charleton, Rebecca C., Griffiths, Bridget, Edwards, Elizabeth A., Partlett, Rosamund, Martin, Keith, Tarzi, Mike, Panthakalam, Sathianathan, Freeman, Tanya, Ainley, Louise, Turner, Michael, Hughes, Lyndsay, Russell, Bridget, Jenkins, Suzanne, Done, John, Young, Adam, Jones, Tadeusz, Gaywood, Ian C., Pande, Ira, Pradère, Marie-Josèphe, Bhaduri, Mahua, Smith, Angela, Cook, Haley, Abraham, Sonya, Ngcozana, Tanaka, Denton, Christopher P., Parker, Louise, Black, Carol M., Ong, Voon, Thompson, Nicola, White, Catherine, Duddy, Martin, Jobanputra, Paresh, Bacon, Pauline, Smith, Jacqueline, Richardson, Ann, Giancola, Giorgio, Soh, Vicky, Spencer, Simon, Greenhalgh, Ann, Hanson, Mark, De Lord, Denise, Lloyd, Mark, Wong, Henna, Wren, Damian, Grover, Bob, Hall, John, Neville, Cai, Alton, Peter, Kelly, Stephen, Bombardieri, Michele, Humby, Fran, Ng, Nora, Di Cicco, Maria, Hands, Rebecca, Epis, Oscar, Filer, Andrew, Buckley, Christopher, McInnes, Iain, Taylor, Peter, Pitzalis, Costantino, Freeston, Jane, Conaghan, Philip, Grainger, Andrew, O'Connor, Philip J., Evans, Rob, Emery, Paul, Hodgson, Richard, Fleischmann, Roy, Han, Chenglong, van der Heijde, Desiree, Xu, Weichun, Hsia, Elizabeth, Kavanaugh, Arthur, Gladman, Dafna, Chattopadhyay, Chandrabhusan, Beutler, Anna, Zayat, Ahmed S., Ellegard, Karen, Terslev, Lene, Wakefield, Richard J., Ciurtin, Coziana, Leandro, Maria, Dey, Dzifa, Nandagudi, Anupama, Giles, Ian, Shipley, Mike, Morris, Vanessa, Ioannou, John, Ehrenstein, Michael, Sen, Debajit, Chan, Marian, Quinlan, Tim M., Brophy, Richard, Mewar, Devesh, Patel, Dipti, Wilby, M. J., Pellegrini, Vincent, Eyes, Brian, Crooks, Daniel, Anderson, Marina, Ball, Elisabeth, McKeeman, Helen, Burns, James, Yau, Wing Hoi, Moore, Owen, Foo, Joanne, Benson, Claire, Patterson, Chris, Wright, Gary, Taggart, Allister, Drew, Sarah, Tanner, Lorraine, Sanyal, Kaushik, Bourke, Brian E., Alston, Claire, Baqai, Charlotte, Chard, Michael, Sandhu, Virinderjit, Jordan, Kelsey, Munns, Catherine, Zouita, Louisa, Shattles, Warren, Davies, Ursula, Makadsi, Raad, Griffith, Sian, Kiely, Patrick D., Dimofte, Ionela, Dabu, Mihaela, Dabu, Bogdan, Dobarro, David, Schreiber, Benjamin E., Warrell, Clare, Handler, Clive, Coghlan, Gerry, Denton, Chris, Ishorari, Jasmine, Bunn, Chris, Beynon, Huw, George Malal, Joby J., Boton-Maggs, Ben, Leung, Alexander, Farewell, Daniel, Choy, Ernest, Gullick, Nicola J., Choy, Ernest H., Wincup, Chris, Fisher, Ben, Charles, Peter, Pollard, Louise C., Kirkham, Bruce W., Ma, Margaret H., Ramanujan, Saroja, Cavet, Guy, Haney, Doug, Kingsley, Gabrielle H., Scott, David, Cope, Andrew, Singh, Animesh, Wilson, Jo, Isaacs, Anthony, Wing, Charlotte, McLaughlin, Maeve, Penn, Henry, Genovese, Mark C., Sebba, Anthony, Rubbert-Roth, Andrea, Scali, Juan, Zilberstein, Moshe, Thompson, Liz, Van Vollenhoven, Ronald, De Benedetti, Fabrizio, Brunner, Hermine, Allen, Roger, Brown, Diane, Chaitow, Jeffrey, Pardeo, Manuela, Espada, Graciela, Flato, Berit, Horneff, Gerd, Devlin, Clare, Kenwright, Andrew, Schneider, Rayfel, Woo, Patricia, Martini, Alberto, Lovell, Daniel, Ruperto, Nicola, John, Holly, Hale, Elizabeth D., Treharne, Gareth J., Carroll, Douglas, Mercer, Louise, Low, Audrey, Galloway, James, Watson, Kath, Lunt, Mark, Symmons, Deborah, Davies, Rebecca, Dixon, William, Balarajah, Sharmili, Sandhu, Amrita, Ariyo, Mercy, Rankin, Elizabeth, Sandoo, Aamer, van Zanten, Jet J. Veldhuijzen, Toms, Tracey E., Smith, Jacqueline P., Malik, Saadia, Toberty, Elizabeth, Thalayasingam, Nishanthi, Hamilton, Jennifer, Kelly, Clive, Puntis, Daniel, Saravanan, Vadivelu, Rynne, Martin, Heycock, Carol, Winter, Richard, Wardle, Phil, Toms, Tracey, Cadman, Susan, Nightingale, Peter, Kitas, George, Alhusain, Awal Z., Verstappen, Suzanne M., Mirjafari, Hoda, Charlton-Menys, Valentine, Bunn, Diane, Durrington, Paul, Cooney, Jennifer K., Moore, Jonathan P., Lemmey, Andrew, Jones, Jeremy G., Maddison, Peter J., Ahmad, Yasmeen A., Ahmed, Tazeen J., Leone, Francesca, Browne, Hannah K., Wig, Surabhi, Chevance, Aurelie, Moore, Tonia, Manning, Joanne, Vail, Andy, Herrick, Ariane L., Derrett-Smith, Emma, Hoyles, Rachel, Moinzadeh, Pia, Chighizola, Cecilia, Khan, Korsa, Abraham, David, Warrell, Clare E., Sykes, Rebecca, Muir, Lindsay, Ennis, Holly, Shiwen, Xu, Thompson, Katherine, Liu, Shangxi, Leask, Andrew, Abraham, David J., Strickland, Gemma, Pauling, John, Betteridge, Zoe, Dunphy, Juliet, Owen, Pat, McHugh, Neil, Abignano, Giuseppina, Cuomo, Giovanna, Buch, Maya H., Rosenberg, William M., Valentini, Gabriele, Del Galdo, Francesco, Jenkins, Jessica, Pauling, John D., Howell, Kevin, Nihtyanova, Svetlana, Fonseca, Carmen, Malaviya, Anshuman P., Hadjinicolaou, Andreas V., Nisar, Muhammad K., Ruddlesden, Michael, Furlong, Anita, Baker, Sharon, Hall, Frances C., Raut-Roy, Dominique, Peluso, Rosario, Dario Di Minno, M. N., Iervolino, Salvatore, Costa, Luisa, Atteno, Mariangela, Lofrano, Mariana, Soscia, Ernesto, Castiglione, Fabiana, Foglia, Francesca, Scarpa, Raffaele, Wallis, Dinny, Dougados, Maxime, Keystone, Edward, Heckaman, Michele, Mease, Philip, Landewé, Robert, Nguyen, Dianne, Winfield, Rachel A., Dyke, Claire, Clemence, Mark, Mackay, Kirsten, Haywood, Kirstie L., Packham, Jon, Jordan, Kelvin P., Davies, Helen, Brophy, Sinead, Irvine, Elizabeth, Cooksey, Roxanne, Dennis, Michael S., Siebert, Stefan, Ibrahim, Fowzia, Krueger, Gerald, Gathany, Tim, Mudivarthy, Surekha, Mack, Michael, Tandon, Neeta, Sieper, Joachim, Braun, Jürgen, van der Heijde, Désirée, Isaacs, John, Dahmen, Georg, Wollenhaupt, Jürgen, Schulze-Koops, Hendrik, Gsteiger, Sandro, Bertolino, Arthur, Hueber, Wolfgang, Tak, Paul P., Cohen, Carla J., Karaderi, Tugce, Pointon, Jennifer J., Wordsworth, Bryan P., Keidel, Sarah, Farrar, Claire, Appleton, Louise H., Adshead, Rebecca, Tahir, Hasan, Greenwood, Mandy, Donnelly, Simon P., Wajed, Julekha, and Kirkham, Bruce

Abstract

Background: Foot surgery is common in patients with RA but research into surgical outcomes is limited and conceptually flawed as current outcome measures lack face validity: to date no one has asked patients what is important to them. This study aimed to determine which factors are important to patients when evaluating the success of foot surgery in RA Methods: Semi structured interviews of RA patients who had undergone foot surgery were conducted and transcribed verbatim. Thematic analysis of interviews was conducted to explore issues that were important to patients. Results: 11 RA patients (9 ♂, mean age 59, dis dur = 22yrs, mean of 3 yrs post op) with mixed experiences of foot surgery were interviewed. Patients interpreted outcome in respect to a multitude of factors, frequently positive change in one aspect contrasted with negative opinions about another. Overall, four major themes emerged. Function: Functional ability & participation in valued activities were very important to patients. Walking ability was a key concern but patients interpreted levels of activity in light of other aspects of their disease, reflecting on change in functional ability more than overall level. Positive feelings of improved mobility were often moderated by negative self perception ("I mean, I still walk like a waddling duck”). Appearance: Appearance was important to almost all patients but perhaps the most complex theme of all. Physical appearance, foot shape, and footwear were closely interlinked, yet patients saw these as distinct separate concepts. Patients need to legitimize these feelings was clear and they frequently entered into a defensive repertoire ("it's not cosmetic surgery; it's something that's more important than that, you know?”). Clinician opinion: Surgeons' post operative evaluation of the procedure was very influential. The impact of this appraisal continued to affect patients' lasting impression irrespective of how the outcome compared to their initial goals ("when he'd done it ... he said that hasn't worked as good as he'd wanted to ... but the pain has gone”). Pain: Whilst pain was important to almost all patients, it appeared to be less important than the other themes. Pain was predominately raised when it influenced other themes, such as function; many still felt the need to legitimize their foot pain in order for health professionals to take it seriously ("in the end I went to my GP because it had happened a few times and I went to an orthopaedic surgeon who was quite dismissive of it, it was like what are you complaining about”). Conclusions: Patients interpret the outcome of foot surgery using a multitude of interrelated factors, particularly functional ability, appearance and surgeons' appraisal of the procedure. While pain was often noted, this appeared less important than other factors in the overall outcome of the surgery. Future research into foot surgery should incorporate the complexity of how patients determine their outcome Disclosure statement: All authors have declared no conflicts of interest

Published
2017

22. Case Reports1. A Late Presentation of Loeys-Dietz Syndrome: Beware of TGFβ Receptor Mutations in Benign Joint Hypermobility

Author

Mehta, Puja, Holder, Susan, Fisher, Benjamin, Vincent, Tonia, Nadesalingam, Kavitha, Maciver, Helen, Shingler, Wendy, Bakshi, Jyoti, Hassan, Sadon, D'Cruz, David, Chan, Antoni, Litwic, Anna E., McCrae, Fiona, Seth, Rakhi, Nandagudi, Anupama, Jury, Elizabeth, Isenberg, David, Karjigi, Uma, Paul, Anupam, Rees, Frances, O'Dowd, Emma, Kinnear, William, Johnson, Simon, Lanyon, Peter, Stevens, Richard, Narayan, Nehal, Marguerie, Christopher, Robinson, Helena, Ffolkes, Lorrette, Worsnop, Fiona, Ostlere, Lucy, Kiely, Patrick, Dharmapalaiah, Chethana, Hassan, Nada, Bharadwaj, Anurag, Skibinska, Malgorzata, Gendi, Nagui, Davies, Emma J., Akil, Mohammed, Kilding, Rachael, Ramachandran Nair, Jagdish, Walsh, Maeve, Farrar, Wendy, Thompson, Robert N., Borukhson, Liubov, McFadyen, Charles, Singh, Deepwant, Rajagopal, Vivek, Chan, Angela Marie L., Wearn Koh, Li, Christie, Jennifer D., Croot, Lorraine, Gayed, Mary, Disney, Benjamin, Singhal, Saket, Grindulis, Karl, Reynolds, Timothy D., Conway, Katie, Williams, Debbie, Quin, John, Dean, Gillian, Churchill, Duncan, Walker-Bone, Karen E., Goff, Iain, Reynolds, Gary, Grove, Matthew, Patel, Priya, Lazarus, Mark N., Roncaroli, Frederico, Gabriel, Carolyn, Kinderlerer, Anne R., Nikiphorou, Elena, Hall, Frances C., Bruce, Ellen, Gray, Leanne, Krutikov, Maria, Wig, Surabhi, Bruce, Ian, D'Agostino, Maria A., Wakefield, Richard, Berner Hammer, Hilde, Vittecoq, Olivier, Galeazzi, Mauro, Balint, Peter, Filippucci, Emilio, Moller, Ingrid, Iagnocco, Annamaria, Naredo, Esperanza, Ostergaard, Mikkel, Gaillez, Corine, Kerselaers, Wendy, Van Holder, Karina, Le Bars, Manuela, Stone, Millicent A., Williams, Francis, Wolber, Lisa, Karppinen, Jaro, Maatta, Juhani, Thompson, Ben, Atchia, Ismael, Lorenzi, Alice, Raftery, Graham, Platt, Phil, Platt, Philip N., Pratt, Arthur, Turmezei, Thomas D., Treece, Graham M., Gee, Andrew H., Poole, Kenneth E., Chandratre, Priyanka N., Roddy, Edward, Clarson, Lorna, Richardson, Jane, Hider, Samantha, Mallen, Christian, Lieberman, Abigail, Prouse, Peter J., Mahendran, Prini, Samarawickrama, Amanda, Ottery, Faith D., Yood, Robert, Wolfson, Marsha, Ang, Andrea, Riches, Philip, Thomson, Janet, Nuki, George, Humphreys, Jennifer, Verstappen, Suzanne M., Chipping, Jacqueline, Hyrich, Kimme, Marshall, Tarnya, Symmons, Deborah P., Roy, Matthew, Kirwan, John R., Marshall, Robert W., Matcham, Faith, Scott, Ian C., Rayner, Lauren, Hotopf, Matthew, Kingsley, Gabrielle H., Scott, David L., Steer, Sophia, Ma, Margaret H., Dahanayake, Chanaka, Kingsley, Gabrielle, Cope, Andrew, Wernham, Aaron, Ward, Lorna, Carruthers, David, Deeming, Alison, Buckley, Christopher, Raza, Karim, De Pablo, Paola, Carpenter, Lewis, Jayakumar, Keeranur, Solymossy, Csilla, Dixey, Josh, Young, Adam, Singh, Animesh, Penn, Henry, Ellerby, Nicholas, Mattey, Derek L., Packham, Jonathan, Dawes, Peter, Hider, Samantha L., Ng, Nora, Humby, Frances, Bombardieri, Michele, Kelly, Stephen, Di Cicco, Maria, Dadoun, Sabrina, Hands, Rebecca, Rocher, Vidalba, Kidd, Bruce, Pyne, Dev, Pitzalis, Costantino, Poore, Sophie, Hutchinson, David, Low, Audrey, Lunt, Mark, Mercer, Louise, Galloway, James, Davies, Rebecca, Watson, Kath, Dixon, Will, Symmons, Deborah, Watson, Kath D., Dixon, William G., Hyrich, Kimme L., Malik, Saadia P., Kelly, Clive, Hamilton, Jennifer, Heycock, Carol, Saravanan, Vadivelu, Rynne, Martin, Harris, Helen E., Tweedie, Fiona, Skaparis, Yiannis, White, Marie, Scott, Nicola, Samson, Kay, Mercieca, Cecilia, Clarke, Shane, Warner, Alexander J., Verstappen, Suzanne, Chan, Esther, Woodhead, Felix A., Nisar, Mohamed, Arthanari, S., Dawson, Julie, Sathi, Nav, Ahmad, Yasmeen, Koduri, Gouri, Cumming, Jo, Stannett, Peter, Hull, Richard, Metsios, George, Stavropoulos Kalinoglou, Antonios, Veldhuijzen van Zanten, Jet J., Nightingale, Peter, Koutedakis, Yiannis, Kitas, George D., Williams, Peter, Walsh, David, Perry, Elizabeth, de-Soyza, Anthony, Moullaali, Thomas, Eggleton, Paul, Stavropoulos-Kalinoglou, Antonios, Sandoo, Aamer, de Pablo, Paola, Maggs, Fiona, Faizal, Abdul, Pugh, Mark, Jobanputra, Paresh, Kehoe, Oksana, Cartwright, Alison, Askari, Ayman, El Haj, Alicia, Middleton, Jim, Aynsley, Sarah, Hardy, Jacob, Veale, Douglas, Fearon, Ursula, Wilson, Gerry, Muthana, Munitta, Fossati, G., Healy, L., Nesbitt, A., Becerra, Elena, Leandro, Maria J., De La Torre, Inmaculada, Cambridge, Geraldine, Nelson, P. N., Roden, D., Shaw, M., Davari Ejtehadi, Hora, Nevill, A., Freimanis, G., Hooley, P., Bowman, S., Alavi, A., Axford, J., Veitch, A. M., Tugnet, N., Rylance, P. B., Hawtree, Sarah, Mark Wilkinson, J., Wilson, Anthony G., Woon Kam, Ngar, Filter, Andrew, Croft, Adam P., Naylor, Amy, Zimmermann, Birgit, Hardie, Debbie, Desanti, Guillaume, Jaurez, Maria, Muller-Ladner, Ulf, Filer, Andrew, Neumann, Elena, Movahedi, Mohammad, Ray, David W., Burmester, Gerd R., Matucci-Cerinic, Marco, Navarro-Blasco, Francisco, Kary, Sonja, Unnebrink, Kristina, Kupper, Hartmut, Mukherjee, Sandeep, Cornell, Patricia, Richards, Selwyn, Rahmeh, Fouz, Thompson, Paul W., Westlake, Sarah L., Javaid, Muhammad K., Batra, Rajbir, Chana, Jasroop, Round, Gemma, Judge, Andrew, Taylor, Peter, Patel, Sanjeev, Cooper, Cyrus, Ravindran, Vinod, Bingham, Clifton O., Weinblatt, Michael E., Mendelsohn, Alan, Kim, Lilianne, Mack, Michael, Lu, Jiandong, Baker, Daniel, Westhovens, Rene, Hewitt, Jamie, Han, Chenglong, Keystone, Edward C., Fleischmann, Roy, Smolen, Josef, Emery, Paul, Genovese, Mark, Doyle, Mittie, Hsia, Elizabeth C., Hart, Jennifer C., Harland, Dave, Gibbons, Carl, Pang, Hok, Huertas, Catherine, Diamantopoulos, Alex, Dejonckheere, Fred, Clowse, M., Wolf, D., Stach, C., Kosutic, G., Williams, S., Terpstra, I., Mahadevan, U., Ferraccioli, G., Samborski, W., Berenbaum, F., Davies, O., Koetse, W., Bennett, B., Burkhardt, H., Luijtens, K., van der Heijde, Desiree, Mariette, X., van Vollenhoven, Ronald F., Bykerk, V., de Longueville, M., Arendt, C., Cush, J., Khan, Afsha, Maclaren, Zoe, Dubash, Sayam, Chalam, Venkat C., Sheeran, Tom, Price, Tom, Baskar, Sangeetha, Mulherin, Diarmuid, Molloy, Cauline, Keay, Fiona, Heritage, Caroline, Douglas, Barbara, Schiff, Michael H., Khanna, Dinesh, Furst, Daniel E., Maldonado, Michael A., Li, Wanying, Sasso, Eric H., Emerling, Daniel, Cavet, Guy, Ford, Kerri, Mackenzie-Green, Bronwen, Collins, David, Price, Elizabeth, Williamson, Lyn, Golla, Janardhana, Vagadia, Vipul, Morrison, Elaine, Tierney, Ann, Wilson, Hilary, Hunter, John, Reddy, Venkat, Moore, Samantha, Ehrenstein, Michael, Benson, Claire, Wray, Maria, Cairns, Andrew, Wright, Gary, Pendleton, Adrian, McHenry, Michelle, Taggart, Allister, Bell, Aubrey, Bosworth, Ailsa, Cox, Maureen, Johnston, Graeme, Shah, Preeti, O'Brien, Anne, Jones, Peter, Sargeant, Ify, Bukhari, Marwan, Nusslein, Hubert, Alten, Rieke, Lorenz, Hannes M., Boumpas, Dimitrios, Nurmohamed, Michael T., Bensen, William, Peter, Hans-Hartmut, Rainer, Franz, Pavelka, Karel, Chartier, Melanie, Poncet, Coralie, Rauch, Christiane, Lempp, Heidi, Hofmann, Darija, Adu, Aderonke, Congreve, Carron, Dobson, Joanne, Rose, Diana, Simpson, Carol, Wykes, Til, Ibrahim, Fowzia, Schiff, Michael, Nash, Peter, Durez, Patrick, Kaine, Jeffrey, Delaet, Ingrid, Kelly, Sheila, Maldonado, Michael, Patel, Salil, Jones, Graeme, Sebba, Anthony, Lepley, Denise, Devenport, Jenny, Bernasconi, Corrado, Smart, Devi, Mpofu, Chiedzo, Gomez-Reino, Juan J., Verma, Inderjeet, Kaur, Jaspreet, Syngle, Ashit, Krishan, Pawan, Vohra, Kanchan, Kaur, Ladbans, Garg, Nidhi, Chhabara, Monica, Gibson, Kellie, Woodburn, James, Telfer, Scott, Buckley, Felicity, Finckh, Axel, Huizinga, Tom W., Jansen, Jeroen P., Rubbert-Roth, Andrea, Scali, Juan J., Kremer, Joel M., Pitts, Laura, Vernon, Emma, Sharif, Mohammed I., Das, Sudipto, Helliwell, Philip, Sokoll, Katharina, and Vital, Edward M.

Subjects
cardiovascular system
Abstract

Background: Thoracic aortic aneurysms (TAA) and dissections are not uncommon causes of sudden death in young adults. Loeys-Dietz syndrome (LDS) is a rare, recently described, autosomal dominant, connective tissue disease characterized by aggressive arterial aneurysms, resulting from mutations in the transforming growth factor beta (TGFβ) receptor genes TGFBR1 and TGFBR2. Mean age at death is 26.1 years, most often due to aortic dissection. We report an unusually late presentation of LDS, diagnosed following elective surgery in a female with a long history of joint hypermobility. Methods: A 51-year-old Caucasian lady complained of chest pain and headache following a dural leak from spinal anaesthesia for an elective ankle arthroscopy. CT scan and echocardiography demonstrated a dilated aortic root and significant aortic regurgitation. MRA demonstrated aortic tortuosity, an infrarenal aortic aneurysm and aneurysms in the left renal and right internal mammary arteries. She underwent aortic root repair and aortic valve replacement. She had a background of long-standing joint pains secondary to hypermobility, easy bruising, unusual fracture susceptibility and mild bronchiectasis. She had one healthy child age 32, after which she suffered a uterine prolapse. Examination revealed mild Marfanoid features. Uvula, skin and ophthalmological examination was normal. Results: Fibrillin-1 testing for Marfan syndrome (MFS) was negative. Detection of a c.1270G > C (p.Gly424Arg) TGFBR2 mutation confirmed the diagnosis of LDS. Losartan was started for vascular protection. Conclusions: LDS is a severe inherited vasculopathy that usually presents in childhood. It is characterized by aortic root dilatation and ascending aneurysms. There is a higher risk of aortic dissection compared with MFS. Clinical features overlap with MFS and Ehlers Danlos syndrome Type IV, but differentiating dysmorphogenic features include ocular hypertelorism, bifid uvula and cleft palate. Echocardiography and MRA or CT scanning from head to pelvis is recommended to establish the extent of vascular involvement. Management involves early surgical intervention, including early valve-sparing aortic root replacement, genetic counselling and close monitoring in pregnancy. Despite being caused by loss of function mutations in either TGFβ receptor, paradoxical activation of TGFβ signalling is seen, suggesting that TGFβ antagonism may confer disease modifying effects similar to those observed in MFS. TGFβ antagonism can be achieved with angiotensin antagonists, such as Losartan, which is able to delay aortic aneurysm development in preclinical models and in patients with MFS. Our case emphasizes the importance of timely recognition of vasculopathy syndromes in patients with hypermobility and the need for early surgical intervention. It also highlights their heterogeneity and the potential for late presentation. Disclosures: The authors have declared no conflicts of interest

Published
2017

25. 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

26. 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

27. 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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28. 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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30. The trials of anakinra

Author

Burls, Amanda and Jobanputra, Paresh

Subjects
Rheumatoid arthritis -- Care and treatment, Etanercept -- Research, Clinical trials
Published
2004

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. Adverse drug reactions

Author

Hicks, Wanda E, Jones, Judith K, Diepen, Louise R Van, Roswell, Robert, Homer, Dawn, Jobanputra, Paresh, Jacka, Justin, and Shenfield, Gillian M

Subjects
Drug interactions -- Adverse and side effects, Sulfonamides -- Adverse and side effects, Hypoglycemic sulfonylureas -- Adverse and side effects
Published
2001

34. 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
Full Text
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41. A randomised efficacy and discontinuation study of etanercept versus adalimumab (RED SEA) for rheumatoid arthritis: a pragmatic, unblinded, non-inferiority study of first TNF inhibitor use: outcomes over 2 years

Author

Jobanputra, Paresh, primary, Maggs, Fiona, additional, Deeming, Alison, additional, Carruthers, David, additional, Rankin, Elizabeth, additional, Jordan, Alison C, additional, Faizal, Abdul, additional, Goddard, Carolyn, additional, Pugh, Mark, additional, Bowman, Simon J, additional, Brailsford, Sue, additional, and Nightingale, Peter, additional

Published
2012
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