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Pediatric Rheumatology Online Journal

Federica Vanoni, Roberta Caorsi, Sandra Aeby, Marie Cochard, Jordi Antón, Stefan Berg, Riva Brik, Pavla Dolezalova, Isabelle Koné-Paut, Benedicte Neven, Seza Ozen, Pascal Pillet, Silvia Stojanov, Carine Wouters, Marco Gattorno, Michaël Hofer
BACKGROUND: Diagnosis of Periodic Fever, Aphthous stomatitis, Pharyngitis and Cervical Adenitis (PFAPA) syndrome is currently based on the modified Marshall's criteria, but no validated evidence based classification criteria for PFAPA has been established so far. METHODS: A multistep process, based on the Delphi and Nominal Group Technique was conducted. After 2 rounds of e-mail Delphi survey involving 21 experts in autoinflammation we obtained a list of variables that were discussed in an International Consensus Conference...
September 21, 2018: Pediatric Rheumatology Online Journal
Joanie Sims-Gould, Douglas L Race, Heather Macdonald, Kristin M Houghton, Ciarán M Duffy, Lori B Tucker, Heather A McKay
BACKGROUND: Physical activity is essential for ensuring optimal physical function and fitness in children with juvenile idiopathic arthritis (JIA). Although exercise intervention trials informed current clinical practice, few studies addressed why children with JIA do or do not participate in exercise interventions. We aimed to describe perceived barriers and facilitators to the uptake and adherence to a 6-month home-based exercise intervention for children diagnosed with JIA and their parents...
September 20, 2018: Pediatric Rheumatology Online Journal
Lucio Ventura-Ríos, Enrique Faugier, Laura Barzola, L B De la Cruz-Becerra, Guadalupe Sánchez-Bringas, Andrés Rodríguez García, Rocío Maldonado, Johannes Roth, Cristina Hernández-Díaz
BACKGROUND: Musculoskeletal Ultrasonography (MSUS) is an important tool for the clinical assessment in Juvenile Idiopathic Arthritis (JIA). The objective of this study was to evaluate the reliability of MSUS to detect elementary lesions: synovitis, tenosynovitis, cartilage damage and bone erosions in the wrist and metacarpal (MCP) joints of patients with JIA. METHODS: Thirty children in various subgroups of JIA according to ILAR criteria, were included in this cross-sectional study...
September 17, 2018: Pediatric Rheumatology Online Journal
Casper G Schoemaker, Wineke Armbrust, Joost F Swart, Sebastiaan J Vastert, Jorg van Loosdregt, Anouk Verwoerd, Caroline Whiting, Katherine Cowan, Wendy Olsder, Els Versluis, Rens van Vliet, Marlous J Fernhout, Sanne L Bookelman, Jeannette Cappon, J Merlijn van den Berg, Ellen Schatorjé, Petra C E Hissink Muller, Sylvia Kamphuis, Joke de Boer, Otto T H M Lelieveld, Janjaap van der Net, Karin R Jongsma, Annemiek van Rensen, Christine Dedding, Nico M Wulffraat
BACKGROUND: Research on Juvenile Idiopathic Arthritis (JIA) should support patients, caregivers/parents (carers) and clinicians to make important decisions in the consulting room and eventually to improve the lives of patients with JIA. Thus far these end-users of JIA-research have rarely been involved in the prioritisation of future research. MAIN BODY: Dutch organisations of patients, carers and clinicians will collaboratively develop a research agenda for JIA, following the James Lind Alliance (JLA) methodology...
September 15, 2018: Pediatric Rheumatology Online Journal
Jacqueline K K Sit, Winnie K Y Chan
BACKGROUND: Accumulated damage is an important prognostic factor in systemic lupus erythematous. However, the pattern of disease damage and its risk factors have not been well studied in childhood-onset systemic lupus erythematosus (cSLE) in Asia. The objectives are to evaluate the pattern of damage and to identify the risk factors for accumulated damage in an Asian group of cSLE. METHODS: A retrospective chart review was conducted on a group of 59 patients with cSLE...
September 10, 2018: Pediatric Rheumatology Online Journal
Wenji Chen, Shiyan Mo, Gui Luo, Yanyan Wang, Xiaohu Deng, Jian Zhu, Wei Zhao
BACKGROUND: As one kind of osteochondrodysplasia, progressive pseudorheumatoid dysplasia (PPD) is also known as spondyloepiphyseal dysplasia tarda with progressive arthropathy or arthropathy progressive pseudorheumatoid of childhood. PPD is a very rare disease, especially in China, and has an estimated prevalence of 1/1000000 due to lacking definite prevalence survey. It is an autosomal recessive disorder caused by gene mutation of Wntl inducible signaling pathway protein 3 (WISP3). Its basic pathological change is persistent degeneration and loss of articular cartilage in multiple joints...
September 10, 2018: Pediatric Rheumatology Online Journal
Jessica L Bloom, Jeffrey R Darst, Lori Prok, Jennifer B Soep
BACKGROUND: Henoch-Schonlein Purpura (HSP) is one of the most common vasculitides of childhood, with 10-20 cases per 100,000 children. It frequently occurs following an infectious trigger and involves IgA and C3 deposition in small vessel walls. HSP is characterized by palpable purpura plus IgA deposition on biopsy, arthritis/arthralgia, renal involvement (hematuria and/or proteinuria), and/or abdominal pain. It is not generally recognized as a cause of dilated coronary arteries. CASE PRESENTATION: We describe the first reported case of HSP presenting with dilated coronary arteries...
September 4, 2018: Pediatric Rheumatology Online Journal
Veronica Lundberg, Viveca Lindh, Catharina Eriksson, Solveig Petersen, Eva Eurenius
Following publication of the original article [1], the authors reported an error in the data of their article: one girl was by mistake scored as a boy. The authors have made new analyses of the corrected data. The corrected data and the new analyses are listed in this Correction.
August 22, 2018: Pediatric Rheumatology Online Journal
Megan Cann, Athimalaipet V Ramanan, Andrew Crawford, Andrew D Dick, Sarah L N Clarke, Fatima Rashed, Catherine M Guly
BACKGROUND: There is a paucity of data on the ocular outcomes in paediatric non-infectious uveitis since the introduction of the biologic agents. The purpose of this study was to outline the clinical characteristics of children with non-infectious uveitis and determine the visual outcomes and ocular complication rates in the modern era. METHODS: Children with non-infectious uveitis from January 2011 to December 2015 were identified. Data was collected at baseline, 1, 3, 5, and 10 years post diagnosis...
August 6, 2018: Pediatric Rheumatology Online Journal
Margot Walter, Sylvia Kamphuis, Philomine van Pelt, Annemarie de Vroed, Johanna M W Hazes
BACKGROUND: In 2008 a clinical transition pathway for young people with juvenile-onset rheumatic and musculoskeletal diseases (jRMD) aiming at improving transitional care was instituted. Historical data on drop-out rate in our clinic was 35%, one year before the implementation of the transition pathway. This study aims to I) evaluate the effectiveness of the clinical transition pathway, II) evaluate the experiences and satisfaction of YP with the transitional process and evaluate their perceived self-management skills...
August 3, 2018: Pediatric Rheumatology Online Journal
Ausra Snipaitiene, Rima Sileikiene, Justina Klimaite, Edita Jasinskiene, Rimantas Uktveris, Lina Jankauskaite
BACKGROUND: Chronic recurrent multifocal osteomyelitis (CRMO) is a rare auto-inflammatory bone disorder that primarily affects young girls, with a mean age of 10 years at onset. Generally, it is a self-limited disease. However, recent data indicate that more than 50% of patients have a chronic persistent disease and about 20% a recurring course of this condition. Also, there are more cases reported with associated auto-inflammatory and autoimmune diseases. In this case report, we present a rare case of sporadic CRMO in which the patient eventually developed C-ANCA (cytoplasmic anti-neutrophil cytoplasmic antibodies)-associated renal vasculitis and hyperparathyroidism...
July 27, 2018: Pediatric Rheumatology Online Journal
Dima Chbeir, Jean Gaschignard, Ronan Bonnefoy, Constance Beyler, Isabelle Melki, Albert Faye, Ulrich Meinzer
BACKGROUND: Kawasaki disease (KD) is an acute febrile systemic vasculitis that affects small and medium blood vessels. Intensified treatments for the most severely affected patients have been proposed recently, and the early identification of KD patients at high risk for coronary artery aneurysms (CAA) is crucial. However, the risk scoring systems developed in Japan have not been validated in European populations, and little data is available concerning the link between initial echocardiogram findings other than high z-scores and cardiac prognosis...
July 18, 2018: Pediatric Rheumatology Online Journal
Hanna Rahimi, Marinka Twilt, Troels Herlin, Lynn Spiegel, Thomas Klit Pedersen, Annelise Küseler, Peter Stoustrup
BACKGROUND: Little is known about the chronicity of orofacial symptoms and how this influences the oral health-related quality of life in juvenile idiopathic arthritis (JIA). Therefore, our objectives were to study the long-term changes in self-reported orofacial symptoms, and to define the impact of orofacial symptoms on oral health-related quality of life in JIA. METHODS: At baseline (T0), 157 consecutive JIA patients ≤20 years completed a patient pain questionnaire that incorporates domains related to the orofacial area...
July 13, 2018: Pediatric Rheumatology Online Journal
Giovanna Ferrara, Greta Mastrangelo, Patrizia Barone, Francesco La Torre, Silvana Martino, Giovanni Pappagallo, Angelo Ravelli, Andrea Taddio, Francesco Zulian, Rolando Cimaz
BACKGROUND: Conventional pharmacological therapies for the treatment of juvenile idiopathic arthritis (JIA) consist of non-biological, disease-modifying antirheumatic drugs, among which methotrexate (MTX) is the most commonly prescribed. However, there is a lack of consensus-based clinical and therapeutic recommendations for the use of MTX in the management of patients with JIA. Therefore, the Methotrexate Advice and RecommendAtions on Juvenile Idiopathic Arthritis (MARAJIA) Expert Meeting was convened to develop evidence-based recommendations for the use of MTX in the treatment of JIA...
July 11, 2018: Pediatric Rheumatology Online Journal
Hermine I Brunner, Lisa G Rider, Daniel J Kingsbury, Dominic Co, Rayfel Schneider, Ellen Goldmuntz, Karen B Onel, Edward H Giannini, Daniel J Lovell
IMPORTANCE: Specialized research networks are essential to achieve drug approvals for rare pediatric diseases. Such networks help realize the potential of global legislation enacted upon the recognition that most children are treated with drugs whose most beneficial dose and regimen have not been established in pediatric patients. The Pediatric Rheumatology Collaborative Study Group (PRCSG) is a North American clinical trials network that is specialized in the performance of clinical trials of new therapies for pediatric populations with rheumatic diseases...
July 11, 2018: Pediatric Rheumatology Online Journal
Qianzi Zhao, Lawrence K Jung
BACKGROUND: To understand the relationship between regulatory B cells (Bregs) and juvenile idiopathic arthritis (JIA), we analyzed the percentages of Bregs and their function in peripheral blood (PB) and synovial fluid (SF) of JIA patients. METHODS: Twenty-one JIA patients and 11 children with growing pain but without known rheumatic diseases as controls were included. The B cell phenotype and intracellular production of IL-10 of Bregs were assessed by flow cytometry...
July 4, 2018: Pediatric Rheumatology Online Journal
Fajer Altammar, Bianca Lang
BACKGROUND: Kawasaki Disease (KD), the leading cause of acquired heart disease in children in the developed world, is extremely rare in neonates. We present a case of incomplete KD in a neonate and a review of the literature on neonatal KD. CASE PRESENTATION: A previously healthy full term 15 day old Caucasian male with an unremarkable antenatal and perinatal history, presented on Day 2 of illness with fever, rash, irritability, and poor feeding. Examination revealed fever (39...
July 3, 2018: Pediatric Rheumatology Online Journal
Daniel J Lovell, Jason A Dare, Megan Francis-Sedlak, Julie Ball, Brian D LaMoreaux, Emily Von Scheven, Adam Reinhardt, Rita Jerath, Oral Alpan, Ramesh Gupta, Donald Goldsmith, Andrew Zeft, Henry Naddaf, Beth Gottlieb, Lawrence Jung, Robert J Holt
BACKGROUND: Juvenile idiopathic arthritis (JIA) is an inflammatory arthritis of unknown etiology, which lasts for greater than 6 weeks with onset before 16 years of age. JIA is the most common chronic rheumatic disease in children. NSAIDs have been the mainstay of initial management with naproxen (NAP) being commonly used, but they may cause serious side effects such as gastric ulcers which can be reduced by concomitant administration of proton pump inhibitors, such as esomeprazole (ESO)...
June 26, 2018: Pediatric Rheumatology Online Journal
Claas H Hinze, Prasad T Oommen, Frank Dressler, Andreas Urban, Frank Weller-Heinemann, Fabian Speth, Elke Lainka, Jürgen Brunner, Heike Fesq, Dirk Foell, Wolfgang Müller-Felber, Ulrich Neudorf, Christoph Rietschel, Tobias Schwarz, Ulrike Schara, Johannes-Peter Haas
BACKGROUND: Juvenile dermatomyositis (JDM) is the most common inflammatory myopathy in childhood and a major cause of morbidity among children with pediatric rheumatic diseases. The management of JDM is very heterogeneous. The JDM working group of the Society for Pediatric Rheumatology (GKJR) aims to define consensus- and practice-based strategies in order to harmonize diagnosis, treatment and monitoring of JDM. METHODS: The JDM working group was established in 2015 consisting of 23 pediatric rheumatologists, pediatric neurologists and dermatologists with expertise in the management of JDM...
June 25, 2018: Pediatric Rheumatology Online Journal
Kim Ohl, Helge Nickel, Halima Moncrieffe, Patricia Klemm, Anja Scheufen, Dirk Föll, Viktor Wixler, Angela Schippers, Norbert Wagner, Lucy R Wedderburn, Klaus Tenbrock
BACKGROUND: Inflammatory effector T cells trigger inflammation despite increased numbers of Treg cells in the synovial joint of patients suffering from juvenile idiopathic arthritis (JIA). The cAMP response element (CREM)α is known to play a major role in regulation of T cells in SLE, colitis, and EAE. However, its role in regulation of effector T cells within the inflammatory joint is unknown. METHODS: CREM expression was analyzed in synovial fluid cells from oligoarticular JIA patients by flow cytometry...
June 20, 2018: Pediatric Rheumatology Online Journal
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