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Paediatric Rheumatology European Society Congress: Part Two Genoa, Italy Pediatric Rheumatology 2017, 15(Suppl 1):44 DOI 10.1186/s12969-017-0142-8 MEETINGABSTRACTS Open Access Proceedings of the 23rd Paediatric Rheumatology European Society Congress: part two Genoa, Italy. 28 September – 01 October 2016 Published: 30 May 2017 About this supplement These abstracts have been published as part of Pediatric Rheumatology Volume 15 Supplement 1. The full contents of the supplement are available online at: https://ped-rheum.biomedcentral.com/articles/supplements/volume-15-supplement-1. Please note this is part 2 of 3. glucocorticoids intake reduced, the period between diagnosis verification P178 and immunosuppressants and GIBP prescription decreased. However, it Features of drug therapy of patients with systemic juvenile is still widely used antibiotics, non-selective NSAIDs and glucocorticoids. idiopathic arthritis, according to the Russian register of the Disclosure of Interest Russian union of pediatricians None Declared Olga Lomakina1, Ekaterina Alekseeva1, Sania Valieva1, Tatiana Bzarova1, Irina Nikishina2, Elena Zholobova3, Svetlana Rodionovskaya2, Maria Kaleda2 1Rheumatology, Scientific Center of Children’s Health, Moscow, Russian P179 Federation; 2Rheumatology, V.A. Nasonova Research Institute of Bicipital synovial cyst associated with systemic juvenile idiopathic Rheumatology, Moscow, Russian Federation; 3Rheumatology, Sechenov arthritis: clinical description, sonographic and pathological findings First Moscow State Medical University, Moscow, Russian Federation 1 2 2 2 Yasuo Nakagishi , Masaki Shimizu , Mao Mizuta , Akihiro Yachie Presenting author: Olga Lomakina 1 Department of Pediatric Rheumatology, Hyogo Prefectural Kobe Pediatric Rheumatology 2017, 15(Suppl 1):P178 2 Children’s Hospital, Kobe, Japan; Department of Pediatrics, School of Medicine, Institute of Medical Pharmaceutical and Health Sciences, Introduction: Systemic juvenile arthritis - a rare chronic disease. Register - Kanazawa University, Kanazawa, Japan it's an important tool to monitor the effectiveness and safety of GIBP. Presenting author: Yasuo Nakagishi Objectives: Our aim was to study features of the drug therapy of Pediatric Rheumatology 2017, 15(Suppl 1):P179 children with systemic juvenile idiopathic arthritis (sJIA) Methods: We conducted a retrospective data analysis included in the Introduction: Bicipital synovial cyst is a rare manifestation of Register of sJIA cases, for the period from 2002 to 2015 systemic juvenile idiopathic arthritis (s-JIA). It remains unclear how Results: The indicators of 384 children with sJIA are studied. Prior to bicipital synovial cysts arise. the diagnosis verification, all patients were prescribed to intake anti- Objectives: To describe the presentation and clinical course of bicipi- pyretic agents, 98% —antibiotics. After the diagnosis, non-steroidal tal synovial cysts in 2 patients with s-JIA and to assess how bicipital anti-inflammatory drugs (NSAIDs) were intaken by 282 (73.4%) pa- synovial cysts arise. tients: diclofenac sodium — by 163 (40.1%), nimesulide — by 88 Methods: We report 2 patients with bicipital synovial cyst associated (22.9%) patients. The average duration of NSAID intake from 2002 to with s-JIA. We performed sonographic examinations of bicipital syn- 2015 decreased from 81.5 ± 115.3 to 3.3 ± 3.7 months (p < 0.001). ovial cyst. Furthermore, we investigated pathological examination Prior to the diagnosis verification, glucocorticoids were received using biopsy specimen. intravenously or intramuscularly by 265 (69.0%) patients, orally — Results: Patient 1: Eight-year-old boy was diagnosed as s-JIA at the age 176 (45.8%). Totally, glucocorticoids were received by 330 (85.9%) pa- of 4. His disease course was steroid-dependent and tocilizumab (TCZ) tients: methylprednisolone — 300 of 384 (78.1%), prednisolone — 174 was started from the age of 5. However, his disease relapsed at the age (45.3%), there were totally 1855 prescriptions in 668 cases. The average of 7. He presented with fever and swelling of upper left arm. USG re- duration of glucocorticoid intake from 2002 to 2015 decreased from vealed a hyperechogenic cyst along the margin of the biceps muscle. 13.7 ± 26.7 to 5.0 ± 3,8 months (p < 0.001). As a disease-modifying drug, The biopsy of the cyst revealed cyst was surrounded by granulation tis- methotrexate was intaken by 237 (61.7%), Cyclosporin — by 193 sue with abundant macrophages infiltrate and there were no synovial (50.6%) patients. There were totally 809 cases of genetically engineered tissues. The cyst disappeared with control of disease activity. biological preparations (GIBP) in 430 patients: in 2002–2005–8, in 2011– Patient 2: Twelve-year-old boy was diagnosed as s-JIA at the age of 2015–602 in 397 cases (p = 0.001). Tocilizumab is intaken by 210 8. His disease course was steroid-dependent and tocilizumab (TCZ) (52.9%) of 397 patients, kanakinumab — 37 (9.3%) patients. The disease was started from the age of 12. At the time to start TCZ, he pre- duration from the manifestation to the prescription of immunosuppres- sented with swelling of upper right arm. Ultrasonography revealed a sive drugs from 2002 to 2015 decreased from 27.3 ± 23.9 to1.0 ± hyperechogenic cyst in the fascia of biceps muscle. The biopsy of the 0 months (p < 0.001), GIBP prescriptions — from 70.7 ± 26.3 to 0.5 ± cyst revealed cyst was surrounded by granulation and fibrous tissue 0.7 months, respectively (p < 0.001) with abundant macrophages, lymphocytes and neutrophils infiltrate. Conclusion: In 13 years there have been positive changes in the anti- There were no synovial tissues. The cyst disappeared with control of rheumatic therapy in children with sJIA — the duration of NSAIDs and disease activity. © The Author(s). 2017 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Pediatric Rheumatology 2017, 15(Suppl 1):44 Page 106 of 259 Conclusion: These findings indicate bicipital synovial cysts arise as Table 1 (abstract P180). Summary of the five SJIA patients with MEFV follows: the fluid arises within the shoulder joint and then descends gene mutations into the contiguous bicipital tendon sheath. The tendon eventually Sex Age of MEFV mutations recurrent Effectiveness Other ruptures, leading to collection of fluid in the bicipital area and syn- onset disease of colchicine treatments ovial cysts arise from the biceps muscle fasciitis. Bicipital synovial cyst is a rare manifestation of s-JIA but synovial cyst should be considered case 1 F 6 L110P, E148Q + + TCZ, TAC, PSL in the differential diagnosis in all children with s-JIA presenting with case 2 F 13 L110P, E148Q, + − TCZ, CyA, PSL swelling of the upper arm. P369S, R408Q Disclosure of Interest case 3 F 3 L110P, E148Q, + + TCZ None Declared G304R case 4 F 7 G304R + + TCZ P180 case 5 M 2 E148Q − + TCZ Systemic juvenile idiopathic arthritis with MEFV gene mutations may have a good response to colchicine: suggestion from 5 cases F female, M male, TCZ tocilizumab, TAC tacrolimus, PSL prednisolone, CyA in our center Cyclosporin A Yuko Sugita, Nami Okamoto, Kousuke Shabana, Takuji Murata, Hiroshi Tamai Poster Session: Systemic lupus Osaka Medical and Pharmaceutical University, Takatsuki-city, Japan Presenting author: Yuko Sugita erythematosus and antiphospholipid Pediatric Rheumatology 2017, 15(Suppl 1):P180 syndrome I Introduction: Systemic juvenile idiopathic arthritis (SJIA) is considered P181 to be an autoinflammatory disease in which the dysregulation of Predictors of recovery from lupus nephritis in children innate immune response is suggested to be the main pathogenesis, Eve M. Smith1, Peng Yin2, Andrea L. Jorgensen2, Michael W. Beresford1,3 and there are some reports that show SJIA patients have a significantly on behalf of On behalf of the UK JSLE Cohort Study higher frequency of MEFV gene mutations, which is associated to the 1Women and Children's Health, Institute of Translational Medicine, activation of IL-1β pathway, than healthy control population1,2. University of Liverpool, Liverpool, UK ;2Department of Biostatistics, Objectives: Since the effectiveness of colchicine, which is useful in Institute of Translational Medicine, University of Liverpool, Liverpool, UK; familial Mediterranean fever, in SJIA patients with MEFV gene muta- 3Department of Paediatric Rheumatology, Alder Hey Children's NHS tions is not established, we sought to examine whether colchicine is Foundation Trust, Liverpool, UK effective in such patients. Presenting author: Eve M. Smith Methods: We searched for gene mutations that is responsible for Pediatric Rheumatology 2017, 15(Suppl 1):P181 autoinflammatory disease in SJIA patients who had persistent clinical symptoms (e.g., skin rashes and arthritis) or flare even under treat- Introduction: Within an adult-onset Systemic Lupus Erytematosus ment with glucocorticoid, disease-modifying antirheumatic drugs (SLE) context, proteinuria has been shown to take a significant period (DMARDS), and IL-6 receptor inhibitor (tocilizumab). We obtained of time to normalise, with 53% of lupus
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