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7. 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

8. 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

9. Rheumatoid arthritis - clinical aspects: 134. Predictors of Joint Damage in South Africans with Rheumatoid Arthritis

Author

Ickinger, Claudia, Musenge, Eustasius, Tikly, Mohammed, Barnes, Jonathan, Donnison, C., Scott, M., Bartholomew, Peter, Rynne, Martin, Hamilton, Jennifer, Saravanan, V., Heycock, Carol, Kelly, Clive, de la Torre, Inmaculada, Moura, Rita A., Leandro, Maria, Edwards, Jonathan, Cambridge, Geraldine, Edwards, Johnathan, Daniels, Louise E., Gullick, Nicola J., Rees, Jonathan D., Kirkham, Bruce W., Rees, Jonathan, Scott, Ian C., Johnson, Deborah, Scott, David L., Kingsley, Gabrielle, Ma, Margaret H., Cope, Andrew P., Brode, Sven, Nisar, Muhammad K., Östör, Andrew J., Oakley, Stephen P., Jones, Tim, Mistlin, Alan, Panayi, Gabriel S., El Miedany, Yasser, Palmer, Deborah, Porkodi, Ramanathan, Rajendran, Panchapakesa, Waller, Rosemary, Williamson, Lyn, Collins, David, Price, Elizabeth, Juarez, Maria J., El Gaafary, Maha, Youssef, sally, Cramp, Fiona, Hewlett, Sarah, Almeida, Celia, Kirwan, John, Choy, Ernest, Chalder, Trudie, Pollock, Jon, Christensen, Robin, Mirjafari, Hoda, Verstappen, Suzanne, Bunn, Diane, Edlin, Helena, Charlton-Menys, Valentine, Pemberton, Philip, Marshall, Tarnya, Wilson, Paddy, Lunt, Mark, Symmons, Deborah, Bruce, Ian N., Bell, Carolyn, Rowe, Ian F., Jayakumar, Keeranur, Norton, Sam J., Dixey, Josh, Williams, Peter, Young, Adam, Kurunadalingam, Hariney, Parwaiz, Iram, Kumar, Kanta, Howlett, Kath, Hands, Becki, Raza, Karim, Pitzalis, Costantino, Buckley, Chris, Kelly, Stephen, Filer, Andrew, Wheater, Gill, Hogan, Vanessa E., Onno Teng, YK, Tekstra, Janneke, Tuck, Stephen P., Lafeber, Floris P., Huizinga, Tom W., Bijlsma, Johannes W., Francis, Roger M., Datta, Harish K., van Laar, Jaap, Pratt, Arthur G., Charles, Peter J., Choudhury, Muslima, Wilson, Gill, Venables, Patrick J., Isaacs, John, Stack, Rebecca, Kwiatkowska, Brygida, Rantapaa-Dahlqvist, Solbritt, Saxne, Tore, Sidiropoulos, Prodromos, Kteniadaki, Eleni, Misirlaki, Chariklia, Mann, Herman, Vencovsky, Jiri, Ciurea, Adrian, Tamborrini, Giorgio, Kyburz, Diego, Bastian, Hans, Burmester, Gerd R., Detert, Jacqueline, Buckley, Christopher D., Sheehy, Claire, Shipman, Alexa, Stech, Irina, Mukhtyar, Chetan, Atzeni, Fabiola, Sitia, Simona, Tomasoni, Livio, Gianturco, Luigi, Ricci, Cristian, Sarzi-Puttini, Piercarlo, De Gennaro Colonna, Vito, Turiel, Maurizio, Galloway, James, Low, Audrey, Mercer, Louise K., Dixon, Will, Ustianowski, Andrew, Watson, Kath, Fisher, Benjamin, Plant, Darren, Lundberg, Karin, Barton, Anne, Venables, Patrick, Lorenzi, Alice R., and Platt, Philip N.

Abstract

Background: Rheumatoid arthritis (RA) causes progressive joint damage and functional disability. Studies on factors affecting joint damage as clinical outcome are lacking in Africa. The aim of the present study was to identify predictors of joint damage in adult South Africans with established RA. Methods: A cross-sectional study of 100 black patients with RA of >5 years were assessed for joint damage using a validated clinical method, the RA articular damage (RAAD) score. Potential predictors of joint damage that were documented included socio-demographics, smoking, body mass index (BMI), disease duration, delay in disease modifying antirheumatic drug (DMARD) initiation, global disease activity as measured by the disease activity score (DAS28), erythrocyte sedimentation rate (ESR), C reactive protein (CRP), and autoantibody status. The predictive value of variables was assessed by univariate and stepwise multivariate regression analyses. A p value

Published
2017

10. Rheumatoid arthritis - treatment: 180. Utility of Body Weight Classified Low-Dose Leflunomide in Japanese Rheumatoid Arthritis

Author

Nishikawa, Masataka, Owaki, Hajime, Fuji, Takeshi, Soliman, Moetaza M., Ashcroft, Darren M., Watson, Kath D., Lunt, Mark, Symmons, Deborah, Hyrich, Kimme L., Atkinson, Francesca, Malik, Saadia, Heycock, Carol, Saravanan, Vadivelu, Rynne, Martin, Hamilton, Jennifer, Kelly, Clive, Burmester, Gerd, Kary, Sonja, Unnebrink, Kristina, Guérette, Benoit, Oezer, Uemit, Kupper, Hartmut, Dennison, Elaine, Jameson, Karen, Hyrich, Kimme, Watson, Kath, Landewe, Robert, Keystone, Edward, Smolen, Josef, Goldring, Mary, Patra, Kaushik, Cifaldi, Mary, van der Heijde, Désirée, Lloyd, Lois A., Owen, Catherine, Breslin, Anne, Ahmad, Yasmeen, Emery, Paul, Matteson, E. L., Genovese, M., Sague, S., Hsia, E. C., Doyle, M. K., Fan, H., Elashoff, M., Kirkham, Bruce, Wasco, M. C., Bathon, J., Fleischmann, R., Genovese, M. C., Liu, H., Goldman, J., Leirisalo-Repo, M., Zanetakis, E., El-Kadi, H., Kellner, H., Bolce, R., Wang, J., Dehoratius, R., Decktor, D., Kremer, J., Taylor, Peter, Mendelsohn, A., Baker, D., Kim, L., Ritchlin, C., Mariette, Xavier, Matucci Cerenic, Marco, Pavelka, Karel, van Vollenhoven, Ronald, Heatley, Rebecca, Walsh, Claire, Lawson, Richard, Reynolds, Alan, Iaremenko, Oleg, Mikitenko, Ganna, Kavanaugh, Arthur, Luijtens, Kristel, Curtis, Jeffrey, Schiff, Michael, Kvien, Tore, Khanna, Dinesh, Furst, Dan, Behrens, Frank, Koehm, Michaela, Scharbatke, Eva C., Kleinert, Stefan, Weyer, Gerd, Tony, Hans-Peter, Burkhardt, Harald, Blunn, Karina J., Williams, Richard B., Young, Adam, McDowell, Jackie, Weinblatt, Michael, Haraoui, Boulos, Mozaffarian, Neelufar, Khraishi, Majed, Alten, Rieke, Gomez-Reino, Juan, Rizzo, Warren, Schechtman, Joy, Kahan, Andre, Vernon, Emma, Taylor, Monet, Hogan, Vanessa, Holweg, Cecile, Kummerfeld, Sarah, Teng, Onno, Townsend, Michael, van Laar, Jacob M., Gullick, Nicola J., De Silva, Chulanie, Kirkham, Bruce W., Roy, Sanjoy, Emery, P., van der Heijde, D., Keystone, E., Conaghan, P. G., Xu, W., Baratelle, A., Beutler, A., Rahman, M. U., Nikiphorou, Elena, Kiely, Patrick, Walsh, David A., Williams, Richard, Shah, Devron, Knight, Gregory D., Hutchinson, David G., Dass, Shouvik, Atzeni, Fabiola, Vital, Edward M., Bingham, Sarah J., Buch, Maya, Beirne, Paul, Fleischmann, Roy, Dougados, Maxime, Williams, Sarah, Reynard, Mark, Blackler, Laura, Zain, Amir, Oakley, Stephen, Rees, Jonathon, Jones, Tim, Mistlin, Alan, Panayi, Gabriel, Westhovens, Rene, Durez, Patrick, Genant, Harry, Robles, Manuel, Becker, Jean-Claude, Covucci, Allison, Bathon, Joan, Genovese, Mark C., Luggen, Michael, Le Bars, Manuela, Aranda, Richard, Li, Tracy, Elegbe, Ayanbola, Fichtner, Andreas, Strand, Vibeke, Vencovsky, Jiri, Davies, Rebecca, Galloway, James, Hochberg, Marc, Kelly, Sheila, Khan, Nadar, Qi, Keqin, Pappu, Ramesh, Delaet, Ingrid, Luo, Allison, Torbeyns, Anne, Moreland, Larry, Cohen, Roger, Gujrathi, Sheila, Bykerk, Vivian P., Álvaro-Gracia, Jose, Andres Roman Ivorra, Jose, Nurmohamed, Michael T., Bernasconi, Corrado, Stancati, Andrea, Sibilia, Jean, Östör, Andrew, Strangfeld, Anja, Eveslage, Maria, Listing, Joachim, Herzer, Peter, Liebhaber, Anke, Krummel-Lorenz, Brigitte, Zink, Angela, Breedveld, Ferdinand C., Cohen, Stanley B., Matteson, Eric L., Burke, Laura, Chai, Akiko, Reiss, William, Sweetser, Marianne, Shaw, Tim, Ellis, Shawn D., Ehrenstein, Michael R., Notley, Clare A., Yazici, Yusuf, Ince, Akgun, Baraf, Herbert, Malamet, Raymond, Chung, Carol Y., Hughes, Chris, Faurholm, Bjarne, Dell'Accio, Francesco, Manzo, Antonio, Seed, Michael, Eltawil, Noha, Marrelli, Alessandra, Gould, David, Subang, Christina, Al-Kashi, Adam, De Bari, Cosimo, Winyard, Paul, Chernajovsky, Yuti, Nissim, Ahuva, Bingham, Clifton, Fleischmann, Roy M., Furst, Daniel E., Macey, Katherine M., Sweetser, Marianne T., Lehane, Patricia, Farmer, Pamela, Long, Simon G., Kremer, Joel M., Burgos-Vargas, Ruben, Dudler, Jean, Mela, Christopher M., Wegner, Natalia, Lugli, Helena, Quirke, Anne-Marie, Guo, Yonghua, Potempa, Jan, Venables, Patrick, Nishikawa, Masataka, Owaki, Hajime, Fuji, Takeshi, Soliman, Moetaza M., Ashcroft, Darren M., Watson, Kath D., Lunt, Mark, Symmons, Deborah, Hyrich, Kimme L., Atkinson, Francesca, Malik, Saadia, Heycock, Carol, Saravanan, Vadivelu, Rynne, Martin, Hamilton, Jennifer, Kelly, Clive, Burmester, Gerd, Kary, Sonja, Unnebrink, Kristina, Guérette, Benoit, Oezer, Uemit, Kupper, Hartmut, Dennison, Elaine, Jameson, Karen, Hyrich, Kimme, Watson, Kath, Landewe, Robert, Keystone, Edward, Smolen, Josef, Goldring, Mary, Patra, Kaushik, Cifaldi, Mary, van der Heijde, Désirée, Lloyd, Lois A., Owen, Catherine, Breslin, Anne, Ahmad, Yasmeen, Emery, Paul, Matteson, E. L., Genovese, M., Sague, S., Hsia, E. C., Doyle, M. K., Fan, H., Elashoff, M., Kirkham, Bruce, Wasco, M. C., Bathon, J., Fleischmann, R., Genovese, M. C., Liu, H., Goldman, J., Leirisalo-Repo, M., Zanetakis, E., El-Kadi, H., Kellner, H., Bolce, R., Wang, J., Dehoratius, R., Decktor, D., Kremer, J., Taylor, Peter, Mendelsohn, A., Baker, D., Kim, L., Ritchlin, C., Mariette, Xavier, Matucci Cerenic, Marco, Pavelka, Karel, van Vollenhoven, Ronald, Heatley, Rebecca, Walsh, Claire, Lawson, Richard, Reynolds, Alan, Iaremenko, Oleg, Mikitenko, Ganna, Kavanaugh, Arthur, Luijtens, Kristel, Curtis, Jeffrey, Schiff, Michael, Kvien, Tore, Khanna, Dinesh, Furst, Dan, Behrens, Frank, Koehm, Michaela, Scharbatke, Eva C., Kleinert, Stefan, Weyer, Gerd, Tony, Hans-Peter, Burkhardt, Harald, Blunn, Karina J., Williams, Richard B., Young, Adam, McDowell, Jackie, Weinblatt, Michael, Haraoui, Boulos, Mozaffarian, Neelufar, Khraishi, Majed, Alten, Rieke, Gomez-Reino, Juan, Rizzo, Warren, Schechtman, Joy, Kahan, Andre, Vernon, Emma, Taylor, Monet, Hogan, Vanessa, Holweg, Cecile, Kummerfeld, Sarah, Teng, Onno, Townsend, Michael, van Laar, Jacob M., Gullick, Nicola J., De Silva, Chulanie, Kirkham, Bruce W., Roy, Sanjoy, Emery, P., van der Heijde, D., Keystone, E., Conaghan, P. G., Xu, W., Baratelle, A., Beutler, A., Rahman, M. U., Nikiphorou, Elena, Kiely, Patrick, Walsh, David A., Williams, Richard, Shah, Devron, Knight, Gregory D., Hutchinson, David G., Dass, Shouvik, Atzeni, Fabiola, Vital, Edward M., Bingham, Sarah J., Buch, Maya, Beirne, Paul, Fleischmann, Roy, Dougados, Maxime, Williams, Sarah, Reynard, Mark, Blackler, Laura, Zain, Amir, Oakley, Stephen, Rees, Jonathon, Jones, Tim, Mistlin, Alan, Panayi, Gabriel, Westhovens, Rene, Durez, Patrick, Genant, Harry, Robles, Manuel, Becker, Jean-Claude, Covucci, Allison, Bathon, Joan, Genovese, Mark C., Luggen, Michael, Le Bars, Manuela, Aranda, Richard, Li, Tracy, Elegbe, Ayanbola, Fichtner, Andreas, Strand, Vibeke, Vencovsky, Jiri, Davies, Rebecca, Galloway, James, Hochberg, Marc, Kelly, Sheila, Khan, Nadar, Qi, Keqin, Pappu, Ramesh, Delaet, Ingrid, Luo, Allison, Torbeyns, Anne, Moreland, Larry, Cohen, Roger, Gujrathi, Sheila, Bykerk, Vivian P., Álvaro-Gracia, Jose, Andres Roman Ivorra, Jose, Nurmohamed, Michael T., Bernasconi, Corrado, Stancati, Andrea, Sibilia, Jean, Östör, Andrew, Strangfeld, Anja, Eveslage, Maria, Listing, Joachim, Herzer, Peter, Liebhaber, Anke, Krummel-Lorenz, Brigitte, Zink, Angela, Breedveld, Ferdinand C., Cohen, Stanley B., Matteson, Eric L., Burke, Laura, Chai, Akiko, Reiss, William, Sweetser, Marianne, Shaw, Tim, Ellis, Shawn D., Ehrenstein, Michael R., Notley, Clare A., Yazici, Yusuf, Ince, Akgun, Baraf, Herbert, Malamet, Raymond, Chung, Carol Y., Hughes, Chris, Faurholm, Bjarne, Dell'Accio, Francesco, Manzo, Antonio, Seed, Michael, Eltawil, Noha, Marrelli, Alessandra, Gould, David, Subang, Christina, Al-Kashi, Adam, De Bari, Cosimo, Winyard, Paul, Chernajovsky, Yuti, Nissim, Ahuva, Bingham, Clifton, Fleischmann, Roy M., Furst, Daniel E., Macey, Katherine M., Sweetser, Marianne T., Lehane, Patricia, Farmer, Pamela, Long, Simon G., Kremer, Joel M., Burgos-Vargas, Ruben, Dudler, Jean, Mela, Christopher M., Wegner, Natalia, Lugli, Helena, Quirke, Anne-Marie, Guo, Yonghua, Potempa, Jan, and Venables, Patrick

Abstract

Background: In Japan, more than 20 rheumatoid arthritis (RA) patients died of interstitial pneumonia (IP) caused by leflunomide (LEF) were reported, but many of them were considered as the victims of opportunistic infection currently. In this paper, efficacy and safety of low-dose LEF classified by body weight (BW) were studied. Methods: Fifty-nine RA patients were started to administrate LEF from July 2007 to July 2009. Among them, 25 patients were excluded because of the combination with tacrolimus, and medication modification within 3 months before LEF. Remaining 34 RA patients administered 20 to 50 mg/week of LEF were followed up for 1 year and enrolled in this study. Dose of LEF was classified by BW (50 mg/week for over 50 kg, 40 mg/week for 40 to 50 kg and 20 to 30 mg/week for under 40 kg). The average age and RA duration of enrolled patients were 55.5 years old and 10.2 years. Prednisolone (PSL), methotrexate (MTX) and etanercept were used in 23, 28 and 2 patients, respectively. In case of insufficient response or adverse effect, dosage change or discontinuance of LEF were considered. Failure was defined as dosages up of PSL and MTX, or dosages down or discontinuance of LEF. Last observation carried forward method was used for the evaluation of failed patients at 1 year. Results: At 1 year after LEF start, good/ moderate/ no response assessed by the European League Against Rheumatism (EULAR) response criteria using Disease Activity Score, including a 28-joint count (DAS28)-C reactive protein (CRP) were showed in 14/ 10/ 10 patients, respectively. The dosage changes of LEF at 1 year were dosage up: 10, same dosage: 5, dosage down: 8 and discontinuance: 11 patients. The survival rate of patients in this study was 23.5% (24 patients failed) but actual LEF continuous rate was 67.6% (11 patients discontinued) at 1 year. The major reason of failure was liver dysfunction, and pneumocystis pneumonia was occurred in 1 patient resulted in full recovery. One patient die

Published
2017

18. Hypoadrenalism in patients with fatigue and rheumatic disease.

Author

Methiniti, Dimitra, Hamilton, Jennifer, Saravanan, Vadivelu, Heycock, Carol, and Kelly, Clive

Subjects
DERMATOMYOSITIS, FATIGUE (Physiology), RHEUMATISM, ANEMIA, HYPOTHYROIDISM, STEROID drugs, HYDROCORTISONE, SYSTEMIC lupus erythematosus, AUTOIMMUNE diseases
Abstract

Many patients with rheumatic disease complain of fatigue. Clinicians may interpret this as part of the disease process in the absence of anaemia or hypothyroidism, and sometimes respond with the empiric addition of steroids to therapy. The possibility of true hypoadrenalism is only occasionally considered, and little data exists on the frequency with which it coexists with rheumatic disease. Random serum cortisol may be requested by clinicians to help exclude hypoadrenalism as a factor in fatigue. We undertook a survey to assess how frequently this test was of clinical relevance, what was done in patients with low results, and which patients were most likely to have true adrenal failure. All random cortisol assays requested by the members of a rheumatological team over one year were identified and the notes examined retrospectively. The indication for the request, the result, the ultimate clinical diagnosis and all prior diagnoses were recorded. Where further investigations were undertaken, these too were noted. The results were compared to those in an age and gender matched population of patients with general medical conditions (excluding endocrine disorders) for whom cortisol assays had also been requested. Random cortisol was requested by a team of four consultants in 74 patients with a variety of rheumatic disorders over 12 months, usually because of unexplained fatigue. Among the control group of 75 medical patients, the commonest reasons for requesting cortisol assay were fatigue, low sodium and unexplained anaemia. Mean cortisol levels were significantly higher in medical patients (512 nmol/L) than those with rheumatic disease (351 nmol/L) [P=0.04]. The results were low (<200 nmol/L) in 14 rheumatic patients and 7 medical patients. Among these 21 individuals, synacthen tests were performed in 16 and were indicative of hypoadrenalism in 6 cases. Further investigations revealed primary hypoadrenalism in 3 patients, with tertiary adrenal suppression from oral steroids in three others. All six of these patients had underlying rheumatic rheumatic disorders, usually RA (3) or systemic lupus erythematosus (SLE) (2). None of the medical patients had an abnormal synacthen test. Fatigue in patients with rheumatic disease may be a presenting feature of hypoadrenalism. Although adrenal failure is rare in medical patients with anaemia, hyponatraemia or fatigue, patients with rheumatic disease and unexplained fatigue merit a random cortisol. If this is low, synacthen testing may be appropriate. Steroids should not be commenced empirically in such patients until hypoadrenalism has been excluded, as a significant minority will have genuine primary adrenal failure, usually due to autoimmune disease. [ABSTRACT FROM AUTHOR]

Published
2009
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24. 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, Vital, Edward M., 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.

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 MR

25. Rheumatoid arthritis - clinical aspects: 134. Predictors of Joint Damage in South Africans with Rheumatoid Arthritis

Author

Ickinger, Claudia, Musenge, Eustasius, Tikly, Mohammed, Barnes, Jonathan, Donnison, C., Scott, M., Bartholomew, Peter, Rynne, Martin, Hamilton, Jennifer, Saravanan, V., Heycock, Carol, Kelly, Clive, de la Torre, Inmaculada, Moura, Rita A., Leandro, Maria, Edwards, Jonathan, Cambridge, Geraldine, Edwards, Johnathan, Daniels, Louise E., Gullick, Nicola J., Rees, Jonathan D., Kirkham, Bruce W., Rees, Jonathan, Scott, Ian C., Johnson, Deborah, Scott, David L., Kingsley, Gabrielle, Ma, Margaret H., Cope, Andrew P., Brode, Sven, Nisar, Muhammad K., Östör, Andrew J., Oakley, Stephen P., Jones, Tim, Mistlin, Alan, Panayi, Gabriel S., El Miedany, Yasser, Palmer, Deborah, Porkodi, Ramanathan, Rajendran, Panchapakesa, Waller, Rosemary, Williamson, Lyn, Collins, David, Price, Elizabeth, Juarez, Maria J., El Gaafary, Maha, Youssef, sally, Cramp, Fiona, Hewlett, Sarah, Almeida, Celia, Kirwan, John, Choy, Ernest, Chalder, Trudie, Pollock, Jon, Christensen, Robin, Mirjafari, Hoda, Verstappen, Suzanne, Bunn, Diane, Edlin, Helena, Charlton-Menys, Valentine, Pemberton, Philip, Marshall, Tarnya, Wilson, Paddy, Lunt, Mark, Symmons, Deborah, Bruce, Ian N., Bell, Carolyn, Rowe, Ian F., Jayakumar, Keeranur, Norton, Sam J., Dixey, Josh, Williams, Peter, Young, Adam, Kurunadalingam, Hariney, Parwaiz, Iram, Kumar, Kanta, Howlett, Kath, Hands, Becki, Raza, Karim, Pitzalis, Costantino, Buckley, Chris, Kelly, Stephen, Filer, Andrew, Wheater, Gill, Hogan, Vanessa E., Onno Teng, YK, Tekstra, Janneke, Tuck, Stephen P., Lafeber, Floris P., Huizinga, Tom W., Bijlsma, Johannes W., Francis, Roger M., Datta, Harish K., van Laar, Jaap, Pratt, Arthur G., Charles, Peter J., Choudhury, Muslima, Wilson, Gill, Venables, Patrick J., Isaacs, John, Stack, Rebecca, Kwiatkowska, Brygida, Rantapaa-Dahlqvist, Solbritt, Saxne, Tore, Sidiropoulos, Prodromos, Kteniadaki, Eleni, Misirlaki, Chariklia, Mann, Herman, Vencovsky, Jiri, Ciurea, Adrian, Tamborrini, Giorgio, Kyburz, Diego, Bastian, Hans, Burmester, Gerd R., Detert, Jacqueline, Buckley, Christopher D., Sheehy, Claire, Shipman, Alexa, Stech, Irina, Mukhtyar, Chetan, Atzeni, Fabiola, Sitia, Simona, Tomasoni, Livio, Gianturco, Luigi, Ricci, Cristian, Sarzi-Puttini, Piercarlo, De Gennaro Colonna, Vito, Turiel, Maurizio, Galloway, James, Low, Audrey, Mercer, Louise K., Dixon, Will, Ustianowski, Andrew, Watson, Kath, Fisher, Benjamin, Plant, Darren, Lundberg, Karin, Barton, Anne, Venables, Patrick, Lorenzi, Alice R., Platt, Philip N., Ickinger, Claudia, Musenge, Eustasius, Tikly, Mohammed, Barnes, Jonathan, Donnison, C., Scott, M., Bartholomew, Peter, Rynne, Martin, Hamilton, Jennifer, Saravanan, V., Heycock, Carol, Kelly, Clive, de la Torre, Inmaculada, Moura, Rita A., Leandro, Maria, Edwards, Jonathan, Cambridge, Geraldine, Edwards, Johnathan, Daniels, Louise E., Gullick, Nicola J., Rees, Jonathan D., Kirkham, Bruce W., Rees, Jonathan, Scott, Ian C., Johnson, Deborah, Scott, David L., Kingsley, Gabrielle, Ma, Margaret H., Cope, Andrew P., Brode, Sven, Nisar, Muhammad K., Östör, Andrew J., Oakley, Stephen P., Jones, Tim, Mistlin, Alan, Panayi, Gabriel S., El Miedany, Yasser, Palmer, Deborah, Porkodi, Ramanathan, Rajendran, Panchapakesa, Waller, Rosemary, Williamson, Lyn, Collins, David, Price, Elizabeth, Juarez, Maria J., El Gaafary, Maha, Youssef, sally, Cramp, Fiona, Hewlett, Sarah, Almeida, Celia, Kirwan, John, Choy, Ernest, Chalder, Trudie, Pollock, Jon, Christensen, Robin, Mirjafari, Hoda, Verstappen, Suzanne, Bunn, Diane, Edlin, Helena, Charlton-Menys, Valentine, Pemberton, Philip, Marshall, Tarnya, Wilson, Paddy, Lunt, Mark, Symmons, Deborah, Bruce, Ian N., Bell, Carolyn, Rowe, Ian F., Jayakumar, Keeranur, Norton, Sam J., Dixey, Josh, Williams, Peter, Young, Adam, Kurunadalingam, Hariney, Parwaiz, Iram, Kumar, Kanta, Howlett, Kath, Hands, Becki, Raza, Karim, Pitzalis, Costantino, Buckley, Chris, Kelly, Stephen, Filer, Andrew, Wheater, Gill, Hogan, Vanessa E., Onno Teng, YK, Tekstra, Janneke, Tuck, Stephen P., Lafeber, Floris P., Huizinga, Tom W., Bijlsma, Johannes W., Francis, Roger M., Datta, Harish K., van Laar, Jaap, Pratt, Arthur G., Charles, Peter J., Choudhury, Muslima, Wilson, Gill, Venables, Patrick J., Isaacs, John, Stack, Rebecca, Kwiatkowska, Brygida, Rantapaa-Dahlqvist, Solbritt, Saxne, Tore, Sidiropoulos, Prodromos, Kteniadaki, Eleni, Misirlaki, Chariklia, Mann, Herman, Vencovsky, Jiri, Ciurea, Adrian, Tamborrini, Giorgio, Kyburz, Diego, Bastian, Hans, Burmester, Gerd R., Detert, Jacqueline, Buckley, Christopher D., Sheehy, Claire, Shipman, Alexa, Stech, Irina, Mukhtyar, Chetan, Atzeni, Fabiola, Sitia, Simona, Tomasoni, Livio, Gianturco, Luigi, Ricci, Cristian, Sarzi-Puttini, Piercarlo, De Gennaro Colonna, Vito, Turiel, Maurizio, Galloway, James, Low, Audrey, Mercer, Louise K., Dixon, Will, Ustianowski, Andrew, Watson, Kath, Fisher, Benjamin, Plant, Darren, Lundberg, Karin, Barton, Anne, Venables, Patrick, Lorenzi, Alice R., and Platt, Philip N.

Abstract

Background: Rheumatoid arthritis (RA) causes progressive joint damage and functional disability. Studies on factors affecting joint damage as clinical outcome are lacking in Africa. The aim of the present study was to identify predictors of joint damage in adult South Africans with established RA. Methods: A cross-sectional study of 100 black patients with RA of >5 years were assessed for joint damage using a validated clinical method, the RA articular damage (RAAD) score. Potential predictors of joint damage that were documented included socio-demographics, smoking, body mass index (BMI), disease duration, delay in disease modifying antirheumatic drug (DMARD) initiation, global disease activity as measured by the disease activity score (DAS28), erythrocyte sedimentation rate (ESR), C reactive protein (CRP), and autoantibody status. The predictive value of variables was assessed by univariate and stepwise multivariate regression analyses. A p value <0.05 was considered significant. Results: The mean (SD) age was 56 (9.8) years, disease duration 17.5 (8.5) years, educational level 7.5 (3.5) years and DMARD lag was 9 (8.8) years. Female to male ratio was 10:1. The mean (SD) DAS28 was 4.9 (1.5) and total RAAD score was 28.3 (12.8). The mean (SD) BMI was 27.2 kg/m2 (6.2) and 93% of patients were rheumatoid factor (RF) positive. More than 90% of patients received between 2 to 3 DMARDs. Significant univariate predictors of a poor RAAD score were increasing age (p = 0.001), lower education level (p = 0.019), longer disease duration (p < 0.001), longer DMARD lag (p = 0.014), lower BMI (p = 0.025), high RF titre (p < 0.001) and high ESR (p = 0.008). The multivariate regression analysis showed that the only independent significant predictors of a higher mean RAAD score were older age at disease onset (p = 0.04), disease duration (p < 0.001) and RF titre (p < 0.001). There was also a negative association between BMI and the mean total RAAD score (p = 0.049). Conclusions: Patie

26. 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, Kirkham, Bruce, 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 ("wh

27. Rheumatoid arthritis - treatment: 180. Utility of Body Weight Classified Low-Dose Leflunomide in Japanese Rheumatoid Arthritis

Author

Nishikawa, Masataka, Owaki, Hajime, Fuji, Takeshi, Soliman, Moetaza M., Ashcroft, Darren M., Watson, Kath D., Lunt, Mark, Symmons, Deborah, Hyrich, Kimme L., Atkinson, Francesca, Malik, Saadia, Heycock, Carol, Saravanan, Vadivelu, Rynne, Martin, Hamilton, Jennifer, Kelly, Clive, Burmester, Gerd, Kary, Sonja, Unnebrink, Kristina, Guérette, Benoit, Oezer, Uemit, Kupper, Hartmut, Dennison, Elaine, Jameson, Karen, Hyrich, Kimme, Watson, Kath, Landewe, Robert, Keystone, Edward, Smolen, Josef, Goldring, Mary, Patra, Kaushik, Cifaldi, Mary, van der Heijde, Désirée, Lloyd, Lois A., Owen, Catherine, Breslin, Anne, Ahmad, Yasmeen, Emery, Paul, Matteson, E. L., Genovese, M., Sague, S., Hsia, E. C., Doyle, M. K., Fan, H., Elashoff, M., Kirkham, Bruce, Wasco, M. C., Bathon, J., Fleischmann, R., Genovese, M. C., Liu, H., Goldman, J., Leirisalo-Repo, M., Zanetakis, E., El-Kadi, H., Kellner, H., Bolce, R., Wang, J., Dehoratius, R., Decktor, D., Kremer, J., Taylor, Peter, Mendelsohn, A., Baker, D., Kim, L., Ritchlin, C., Mariette, Xavier, Matucci Cerenic, Marco, Pavelka, Karel, van Vollenhoven, Ronald, Heatley, Rebecca, Walsh, Claire, Lawson, Richard, Reynolds, Alan, Iaremenko, Oleg, Mikitenko, Ganna, Kavanaugh, Arthur, Luijtens, Kristel, Curtis, Jeffrey, Schiff, Michael, Kvien, Tore, Khanna, Dinesh, Furst, Dan, Behrens, Frank, Koehm, Michaela, Scharbatke, Eva C., Kleinert, Stefan, Weyer, Gerd, Tony, Hans-Peter, Burkhardt, Harald, Blunn, Karina J., Williams, Richard B., Young, Adam, McDowell, Jackie, Weinblatt, Michael, Haraoui, Boulos, Mozaffarian, Neelufar, Khraishi, Majed, Alten, Rieke, Gomez-Reino, Juan, Rizzo, Warren, Schechtman, Joy, Kahan, Andre, Vernon, Emma, Taylor, Monet, Hogan, Vanessa, Holweg, Cecile, Kummerfeld, Sarah, Teng, Onno, Townsend, Michael, van Laar, Jacob M., Gullick, Nicola J., De Silva, Chulanie, Kirkham, Bruce W., Roy, Sanjoy, Emery, P., van der Heijde, D., Keystone, E., Conaghan, P. G., Xu, W., Baratelle, A., Beutler, A., Rahman, M. U., Nikiphorou, Elena, Kiely, Patrick, Walsh, David A., Williams, Richard, Shah, Devron, Knight, Gregory D., Hutchinson, David G., Dass, Shouvik, Atzeni, Fabiola, Vital, Edward M., Bingham, Sarah J., Buch, Maya, Beirne, Paul, Fleischmann, Roy, Dougados, Maxime, Williams, Sarah, Reynard, Mark, Blackler, Laura, Zain, Amir, Oakley, Stephen, Rees, Jonathon, Jones, Tim, Mistlin, Alan, Panayi, Gabriel, Westhovens, Rene, Durez, Patrick, Genant, Harry, Robles, Manuel, Becker, Jean-Claude, Covucci, Allison, Bathon, Joan, Genovese, Mark C., Luggen, Michael, Le Bars, Manuela, Aranda, Richard, Li, Tracy, Elegbe, Ayanbola, Fichtner, Andreas, Strand, Vibeke, Vencovsky, Jiri, Davies, Rebecca, Galloway, James, Hochberg, Marc, Kelly, Sheila, Khan, Nadar, Qi, Keqin, Pappu, Ramesh, Delaet, Ingrid, Luo, Allison, Torbeyns, Anne, Moreland, Larry, Cohen, Roger, Gujrathi, Sheila, Bykerk, Vivian P., Álvaro-Gracia, Jose, Andres Roman Ivorra, Jose, Nurmohamed, Michael T., Bernasconi, Corrado, Stancati, Andrea, Sibilia, Jean, Östör, Andrew, Strangfeld, Anja, Eveslage, Maria, Listing, Joachim, Herzer, Peter, Liebhaber, Anke, Krummel-Lorenz, Brigitte, Zink, Angela, Breedveld, Ferdinand C., Cohen, Stanley B., Matteson, Eric L., Burke, Laura, Chai, Akiko, Reiss, William, Sweetser, Marianne, Shaw, Tim, Ellis, Shawn D., Ehrenstein, Michael R., Notley, Clare A., Yazici, Yusuf, Ince, Akgun, Baraf, Herbert, Malamet, Raymond, Chung, Carol Y., Hughes, Chris, Faurholm, Bjarne, Dell'Accio, Francesco, Manzo, Antonio, Seed, Michael, Eltawil, Noha, Marrelli, Alessandra, Gould, David, Subang, Christina, Al-Kashi, Adam, De Bari, Cosimo, Winyard, Paul, Chernajovsky, Yuti, Nissim, Ahuva, Bingham, Clifton, Fleischmann, Roy M., Furst, Daniel E., Macey, Katherine M., Sweetser, Marianne T., Lehane, Patricia, Farmer, Pamela, Long, Simon G., Kremer, Joel M., Burgos-Vargas, Ruben, Dudler, Jean, Mela, Christopher M., Wegner, Natalia, Lugli, Helena, Quirke, Anne-Marie, Guo, Yonghua, Potempa, Jan, Venables, Patrick, Nishikawa, Masataka, Owaki, Hajime, Fuji, Takeshi, Soliman, Moetaza M., Ashcroft, Darren M., Watson, Kath D., Lunt, Mark, Symmons, Deborah, Hyrich, Kimme L., Atkinson, Francesca, Malik, Saadia, Heycock, Carol, Saravanan, Vadivelu, Rynne, Martin, Hamilton, Jennifer, Kelly, Clive, Burmester, Gerd, Kary, Sonja, Unnebrink, Kristina, Guérette, Benoit, Oezer, Uemit, Kupper, Hartmut, Dennison, Elaine, Jameson, Karen, Hyrich, Kimme, Watson, Kath, Landewe, Robert, Keystone, Edward, Smolen, Josef, Goldring, Mary, Patra, Kaushik, Cifaldi, Mary, van der Heijde, Désirée, Lloyd, Lois A., Owen, Catherine, Breslin, Anne, Ahmad, Yasmeen, Emery, Paul, Matteson, E. L., Genovese, M., Sague, S., Hsia, E. C., Doyle, M. K., Fan, H., Elashoff, M., Kirkham, Bruce, Wasco, M. C., Bathon, J., Fleischmann, R., Genovese, M. C., Liu, H., Goldman, J., Leirisalo-Repo, M., Zanetakis, E., El-Kadi, H., Kellner, H., Bolce, R., Wang, J., Dehoratius, R., Decktor, D., Kremer, J., Taylor, Peter, Mendelsohn, A., Baker, D., Kim, L., Ritchlin, C., Mariette, Xavier, Matucci Cerenic, Marco, Pavelka, Karel, van Vollenhoven, Ronald, Heatley, Rebecca, Walsh, Claire, Lawson, Richard, Reynolds, Alan, Iaremenko, Oleg, Mikitenko, Ganna, Kavanaugh, Arthur, Luijtens, Kristel, Curtis, Jeffrey, Schiff, Michael, Kvien, Tore, Khanna, Dinesh, Furst, Dan, Behrens, Frank, Koehm, Michaela, Scharbatke, Eva C., Kleinert, Stefan, Weyer, Gerd, Tony, Hans-Peter, Burkhardt, Harald, Blunn, Karina J., Williams, Richard B., Young, Adam, McDowell, Jackie, Weinblatt, Michael, Haraoui, Boulos, Mozaffarian, Neelufar, Khraishi, Majed, Alten, Rieke, Gomez-Reino, Juan, Rizzo, Warren, Schechtman, Joy, Kahan, Andre, Vernon, Emma, Taylor, Monet, Hogan, Vanessa, Holweg, Cecile, Kummerfeld, Sarah, Teng, Onno, Townsend, Michael, van Laar, Jacob M., Gullick, Nicola J., De Silva, Chulanie, Kirkham, Bruce W., Roy, Sanjoy, Emery, P., van der Heijde, D., Keystone, E., Conaghan, P. G., Xu, W., Baratelle, A., Beutler, A., Rahman, M. U., Nikiphorou, Elena, Kiely, Patrick, Walsh, David A., Williams, Richard, Shah, Devron, Knight, Gregory D., Hutchinson, David G., Dass, Shouvik, Atzeni, Fabiola, Vital, Edward M., Bingham, Sarah J., Buch, Maya, Beirne, Paul, Fleischmann, Roy, Dougados, Maxime, Williams, Sarah, Reynard, Mark, Blackler, Laura, Zain, Amir, Oakley, Stephen, Rees, Jonathon, Jones, Tim, Mistlin, Alan, Panayi, Gabriel, Westhovens, Rene, Durez, Patrick, Genant, Harry, Robles, Manuel, Becker, Jean-Claude, Covucci, Allison, Bathon, Joan, Genovese, Mark C., Luggen, Michael, Le Bars, Manuela, Aranda, Richard, Li, Tracy, Elegbe, Ayanbola, Fichtner, Andreas, Strand, Vibeke, Vencovsky, Jiri, Davies, Rebecca, Galloway, James, Hochberg, Marc, Kelly, Sheila, Khan, Nadar, Qi, Keqin, Pappu, Ramesh, Delaet, Ingrid, Luo, Allison, Torbeyns, Anne, Moreland, Larry, Cohen, Roger, Gujrathi, Sheila, Bykerk, Vivian P., Álvaro-Gracia, Jose, Andres Roman Ivorra, Jose, Nurmohamed, Michael T., Bernasconi, Corrado, Stancati, Andrea, Sibilia, Jean, Östör, Andrew, Strangfeld, Anja, Eveslage, Maria, Listing, Joachim, Herzer, Peter, Liebhaber, Anke, Krummel-Lorenz, Brigitte, Zink, Angela, Breedveld, Ferdinand C., Cohen, Stanley B., Matteson, Eric L., Burke, Laura, Chai, Akiko, Reiss, William, Sweetser, Marianne, Shaw, Tim, Ellis, Shawn D., Ehrenstein, Michael R., Notley, Clare A., Yazici, Yusuf, Ince, Akgun, Baraf, Herbert, Malamet, Raymond, Chung, Carol Y., Hughes, Chris, Faurholm, Bjarne, Dell'Accio, Francesco, Manzo, Antonio, Seed, Michael, Eltawil, Noha, Marrelli, Alessandra, Gould, David, Subang, Christina, Al-Kashi, Adam, De Bari, Cosimo, Winyard, Paul, Chernajovsky, Yuti, Nissim, Ahuva, Bingham, Clifton, Fleischmann, Roy M., Furst, Daniel E., Macey, Katherine M., Sweetser, Marianne T., Lehane, Patricia, Farmer, Pamela, Long, Simon G., Kremer, Joel M., Burgos-Vargas, Ruben, Dudler, Jean, Mela, Christopher M., Wegner, Natalia, Lugli, Helena, Quirke, Anne-Marie, Guo, Yonghua, Potempa, Jan, and Venables, Patrick

Abstract

Background: In Japan, more than 20 rheumatoid arthritis (RA) patients died of interstitial pneumonia (IP) caused by leflunomide (LEF) were reported, but many of them were considered as the victims of opportunistic infection currently. In this paper, efficacy and safety of low-dose LEF classified by body weight (BW) were studied. Methods: Fifty-nine RA patients were started to administrate LEF from July 2007 to July 2009. Among them, 25 patients were excluded because of the combination with tacrolimus, and medication modification within 3 months before LEF. Remaining 34 RA patients administered 20 to 50 mg/week of LEF were followed up for 1 year and enrolled in this study. Dose of LEF was classified by BW (50 mg/week for over 50 kg, 40 mg/week for 40 to 50 kg and 20 to 30 mg/week for under 40 kg). The average age and RA duration of enrolled patients were 55.5 years old and 10.2 years. Prednisolone (PSL), methotrexate (MTX) and etanercept were used in 23, 28 and 2 patients, respectively. In case of insufficient response or adverse effect, dosage change or discontinuance of LEF were considered. Failure was defined as dosages up of PSL and MTX, or dosages down or discontinuance of LEF. Last observation carried forward method was used for the evaluation of failed patients at 1 year. Results: At 1 year after LEF start, good/ moderate/ no response assessed by the European League Against Rheumatism (EULAR) response criteria using Disease Activity Score, including a 28-joint count (DAS28)-C reactive protein (CRP) were showed in 14/ 10/ 10 patients, respectively. The dosage changes of LEF at 1 year were dosage up: 10, same dosage: 5, dosage down: 8 and discontinuance: 11 patients. The survival rate of patients in this study was 23.5% (24 patients failed) but actual LEF continuous rate was 67.6% (11 patients discontinued) at 1 year. The major reason of failure was liver dysfunction, and pneumocystis pneumonia was occurred in 1 patient resulted in full recovery. One patient die

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