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Boeskorova S, Afonskaya M, Argunova V, Sleptsova P, Leonteva L, Burtseva T, Kostik MM. Ethnic heterogeneity of juvenile arthritis in the Republic of Sakha (Yakutia) related to a high human leukocyte antigen B27 frequency. World J Clin Pediatr 2025; 14:101873. [PMID: 40491744 PMCID: PMC11947874 DOI: 10.5409/wjcp.v14.i2.101873] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/29/2024] [Revised: 01/26/2025] [Accepted: 02/10/2025] [Indexed: 03/18/2025] Open
Abstract
BACKGROUND Prevalence of the main rheumatic diseases in the Republic of Sakha (Yakutia) [RS(Y)], one of the regions of the Russian Federation, differs from the other regions of the Russian Federation due to its ethnic and geographic features. Knowledge regarding the prevalence and structure of juvenile idiopathic arthritis (JIA) allows us to shape the work of the pediatric rheumatology service in the region correctly, and optimize the healthcare system and the need for medications. AIM To describe the epidemiological, demographic, clinical, and laboratory characteristics of children with JIA in the RS(Y) and evaluate the main outcomes. METHODS This retrospective cohort study assessed all the data from the medical histories of the patients (n = 225) diagnosed with JIA (2016-2023) in the Cardiorheumatology Department of the M.E. Nikolaev National Center of Medicine. Pearson's χ² test, Fisher's exact test, Mann-Whitney and Kruskal-Wallis tests were used for statistical analyses. RESULTS The ethnic prevalence of JIA is higher in Sakha than in Russian children at 110.1 per 100000 children and 69.4 per 100000 children, respectively. The prevalence of JIA among boys and girls in Sakha was similar, unlike in Russians, where the number of girls predominated. The JIA categories were as follows: (1) Systemic arthritis: 3.5%; (2) Oligoarthritis (persistent and extended): 33.8%; (3) Rheumatoid factor (RF) (+) polyarthritis: 0.9%; (4) RF (-) polyarthritis: 14.7%; (5) Enthesitis-related arthritis (ERA): 44%; and (6) Psoriatic arthritis: 3.1%. Prevalence of the ERA category was 4.4 times higher in Sakha children, but the prevalence of systemic arthritis was 2.9 times lower compared to Russians (P = 0.0005). The frequency of uveitis was 10.2%, and the frequency of human leukocyte antigen (HLA) B27 was 39.6% in JIA children. Biologic treatment was received by 40.4% of JIA children and 45.3% achieved remission. CONCLUSION Higher JIA prevalence, male and ERA predominance, related to a higher frequency of HLA B27 are typical in RS(Y). These data might improve the pediatric rheumatology health service.
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Affiliation(s)
- Sargylana Boeskorova
- Department of Pediatry and Pediatric Surgery, North-Eastern Federal University named after M.K. Ammosov, Medical Institute, Yakutsk 677000, Republic of Sakha (Yakutia), Russia
- Department of Pediatric Rheumatology, Republican Hospital No. 1 –National Center of Medicine Named After M.E. Nikolaev, Yakutsk 677019, Republic of Sakha (Yakutia), Russia
| | - Marina Afonskaya
- Department of Pediatric Rheumatology, Republican Hospital No. 1 –National Center of Medicine Named After M.E. Nikolaev, Yakutsk 677019, Republic of Sakha (Yakutia), Russia
| | - Vera Argunova
- Department of Pediatric Rheumatology, Republican Hospital No. 1 –National Center of Medicine Named After M.E. Nikolaev, Yakutsk 677019, Republic of Sakha (Yakutia), Russia
| | - Polina Sleptsova
- Department of Pediatric Rheumatology, Republican Hospital No. 1 –National Center of Medicine Named After M.E. Nikolaev, Yakutsk 677019, Republic of Sakha (Yakutia), Russia
| | - Liudmila Leonteva
- Department of Rheumatology, Yakut Science Center for Complex Medical Problems, Yakutsk 677000, Republic of Sakha (Yakutia), Russia
- Department of Hospital Therapy, Occupational Diseases and Clinical Pharmacology, North-Eastern Federal University Named After M.K. Ammosov, Medical Institute, Yakutsk 677000, Republic of Sakha (Yakutia), Russia
| | - Tatiana Burtseva
- Department of Pediatry and Pediatric Surgery, North-Eastern Federal University named after M.K. Ammosov, Medical Institute, Yakutsk 677000, Republic of Sakha (Yakutia), Russia
- Department of The Children Heath Monitoring and Environmental Research, Yakut Science Center of The Complex Medical Problems, Yakutsk 677000, Republic of Sakha (Yakutia), Russia
| | - Mikhail Mikhailovich Kostik
- Department of Hospital Pediatry, Saint-Petersburg State Pediatric Medical University, Saint-Petersburg 194100, Russia
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Min EJ, Lee SH, Jo JY, Ahn JG, Jeong DC. Population-Based Big Data Analysis on Disease Patterns in Patients Identified With Juvenile Idiopathic Arthritis Using National Claims Data. J Korean Med Sci 2025; 40:e113. [PMID: 40491085 DOI: 10.3346/jkms.2025.40.e113] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/22/2024] [Accepted: 11/24/2024] [Indexed: 06/11/2025] Open
Abstract
BACKGROUND This study aimed to identify epidemiologic data, including prevalence and incidence, of patients with juvenile idiopathic arthritis (JIA) in accordance with the diseases diagnostic code and Rare Intractable Disease Registration (RIDR) code from the claims data of the Health Insurance Review and Assessment Service (HIRA), the representative secondary data in Korea. METHODS We conducted a retrospective analysis of the national claims data of the HIRA. The study population included 1,728 patients with JIA who 1) had records with diagnostic codes in the format prescribed by the International Classification of Diseases, 10th Revision M08 and the RIDR code V133 between January 1, 2010, and December 31, 2019, and 2) were aged < 16 years as per the definition of JIA. We categorized the patients by age at diagnosis, annual prevalence, and incidence per 100,000 individuals for the study period. Moreover, the study population was stratified by age and gender for subgroup analysis. RESULTS The overall prevalence rate for the total, male, and female population was 15.9 (14.5-16.8), 15.4 (13.1-17.0), and 16.4 (15.9-16.9) per 100,000 individuals, respectively. Additionally, the overall incidence rate for the same populations was 2.2 (1.8-2.8), 2.3 (1.7-2.9), and 2.1 (1.8-2.6) per 100,000 individuals, respectively. CONCLUSION To the best of our knowledge, this is the first study presenting the epidemiologic data, including prevalence and incidence, of patients with JIA using population-based claims big data in Korea. The results of this study will aid in understanding the current status of JIA in Korea and Asia and in health care planning.
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Affiliation(s)
- Eun Jeong Min
- Department of Medical Life Sciences, College of Medicine, The Catholic University of Korea, Seoul, Korea
| | - Soo Hyun Lee
- Division of Medical, Pfizer Biopharmaceuticals Group, Pfizer Pharmaceuticals Korea Limited, Seoul, Korea
| | - Joo-Young Jo
- Division of Medical, Pfizer Biopharmaceuticals Group, Pfizer Pharmaceuticals Korea Limited, Seoul, Korea
| | - Jong Gyun Ahn
- Department of Pediatrics, Severance Children's Hospital, Yonsei University College of Medicine, Seoul, Korea
- Institute for Immunology and Immunological Diseases, Yonsei University College of Medicine, Seoul, Korea.
| | - Dae Chul Jeong
- Department of Pediatrics, College of Medicine, The Catholic University of Korea, Seoul, Korea.
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Jagzape TB, Pandey P, Silpa T, Pinky S. Pediatric Rheumatological Diseases in a Tertiary Care Hospital of Central India: A Retrospective Clinico-Epidemiological Profile. Cureus 2024; 16:e53327. [PMID: 38435943 PMCID: PMC10906977 DOI: 10.7759/cureus.53327] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 01/30/2024] [Indexed: 03/05/2024] Open
Abstract
Introduction: Infectious diseases account for the major health problem in developing countries like India. Though non-infectious diseases like rheumatological disorders are not very common, the burden of these disorders as a group is high in society due to the huge population size. The rheumatological disorders have varied presentations which may mimic other infectious pathologies leading to a significant time lag in the diagnosis. There is inadequate data on the exact burden of these diseases. The spectrum of rheumatological disorders in developing countries is different as compared to the Western world. Hence this study was carried out with the aim of studying the clinical, epidemiological, and laboratory profile of rheumatological disorders in the pediatric age group in a tertiary care hospital. Methods: It was a retrospective study. Data of patients admitted with the diagnosis of rheumatological disorder in the age group of one month to 15 years during the period from June 2018 to December 2022 were reviewed. Results: A total of 35 patients were identified with 20 being female. The mean age of the patients was 8.42± 3.95 years. The most common disease was juvenile idiopathic arthritis (JIA)- 10(28.57%) with an equal proportion of polyarticular JIA and systemic-onset JIA, followed by systemic lupus erythematosus (SLE) nine (25.71%) and Kawasaki Disease (KD)- eight (22.85%). The commonest presenting complaint was fever followed by a rash, whereas the most common findings were pallor and rash. Anemia was present in 25 (71.42%). C-reactive protein (CRP) and erythrocyte sedimentation rate (ESR) were high in 20 (57.14%) and 22 (62.85%), respectively. Antinuclear antibodies (ANA) were positive in 10 (28.57%) and rheumatoid factor (RA) factor in only one (2.85%) case. Conclusions: The most common rheumatological disorder identified was JIA. Fever and rash were the common presenting complaints. Pallor was the commonest sign whereas anemia was the commonest hematological abnormality.
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Affiliation(s)
- Tushar B Jagzape
- Department of Pediatrics, All India Institute of Medical Sciences, Raipur, Raipur, IND
| | - Priyanka Pandey
- Department of Pediatrics, All India Institute of Medical Sciences, Raipur, Raipur, IND
| | - Turaka Silpa
- Department of Pediatrics, All India Institute of Medical Sciences, Raipur, Raipur, IND
| | - Shirisha Pinky
- Department of Pediatrics, All India Institute of Medical Sciences, Raipur, Raipur, IND
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Bhattad S, Mohite R, Singh N. Growth and development in children with rheumatic diseases: Maintaining a balance between drugs and disease activity. INDIAN JOURNAL OF RHEUMATOLOGY 2022. [DOI: 10.4103/injr.injr_54_22] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/05/2022] Open
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Joghataie P, Salahi S, Hosseini L, Rezaeian N. Brucellosis aortitis: An interesting case. Echocardiography 2021; 38:354-356. [PMID: 33483969 DOI: 10.1111/echo.14979] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/19/2020] [Revised: 01/02/2021] [Accepted: 01/07/2021] [Indexed: 01/16/2023] Open
Abstract
A 24-year-old woman referred to our radiology department with the diagnosis of new positive blood culture brucellosis and severe aortic regurgitation for evaluation of periaortic thickening. Cardiac magnetic resonance imaging elegantly revealed the presence of active aortitis, most probably caused by brucellosis.
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Affiliation(s)
| | - Sarvenaz Salahi
- Rajaie Cardiovascular Medical and Research Center, Iran University of Medical Sciences, Tehran, Iran
| | - Leila Hosseini
- Rajaie Cardiovascular Medical and Research Center, Iran University of Medical Sciences, Tehran, Iran.,North Khorasan University of Medical Sciences, Bojnurd, Iran
| | - Nahid Rezaeian
- Rajaie Cardiovascular Medical and Research Center, Iran University of Medical Sciences, Tehran, Iran
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Kobayashi I, Akioka S, Kobayashi N, Iwata N, Takezaki S, Nakaseko H, Sato S, Nishida Y, Nozawa T, Yamasaki Y, Yamazaki K, Arai S, Nishino I, Mori M. Clinical practice guidance for juvenile dermatomyositis (JDM) 2018-Update. Mod Rheumatol 2020; 30:411-423. [PMID: 31955618 DOI: 10.1080/14397595.2020.1718866] [Citation(s) in RCA: 31] [Impact Index Per Article: 6.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/15/2022]
Abstract
Juvenile dermatomyositis is the most common type of juvenile idiopathic inflammatory myopathy mainly affecting the skin and proximal muscles. We have published the Japanese version of 'Clinical practice guidance for juvenile dermatomyositis (JDM) 2018 'consisting of a review of articles in the field and evidence-informed consensus-based experts' opinion on the treatment strategy in collaboration with The Pediatric Rheumatology Association of Japan and The Japan College of Rheumatology under the financial support by 'Research on rare and intractable diseases, Health and Labor Sciences Research Grants'. This article is a digest version of the Japanese guidance.
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Affiliation(s)
- Ichiro Kobayashi
- Center for Pediatric Allergy and Rheumatology, KKR Sapporo Medical Center, Sapporo, Japan
| | - Shinji Akioka
- Department of Pediatrics, Kyoto Prefectural University of Medicine, Kyoto, Japan
| | - Norimoto Kobayashi
- Department of Pediatrics, Shinshu University School of Medicine, Matsumoto, Japan
| | - Naomi Iwata
- Department of Infection and Immunology, Aichi Children's Health and Medical Center, Obu, Japan
| | | | - Haruna Nakaseko
- Department of Infection and Immunology, Aichi Children's Health and Medical Center, Obu, Japan
| | - Satoshi Sato
- Division of Infectious Disease and Immunology, Saitama Children's Medical Center, Omiya, Japan
| | - Yutaka Nishida
- Department of Pediatrics, School of Medicine, Gunma University, Maebashi, Japan
| | - Tomo Nozawa
- Department of Pediatrics, School of Medicine, Yokohama City University, Yokohama, Japan
| | - Yuichi Yamasaki
- Department of Pediatrics, Kagoshima University Hospital, Kagoshima, Japan
| | - Kazuko Yamazaki
- Department of Pediatrics, Saitama Medical Center, Saitama Medical University, Kawagoe, Japan
| | - Satoru Arai
- Department of Dermatology, St. Luke's International Hospital, Tokyo, Japan
| | - Ichizo Nishino
- Department of Neuromuscular Research, National Institute of Neuroscience, National Center of Neurology and Psychiatry, Tokyo, Japan
| | - Masaaki Mori
- Lifetime Clinical Immunology, Tokyo Medical and Dental University, Tokyo, Japan
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Zhang-Jian SJ, Yang HY, Chiu MJ, Chou IJ, Kuo CF, Huang JL, Yeh KW, Wu CY. Pregnancy outcomes and perinatal complications of Asian mothers with juvenile idiopathic arthritis - a case-control registry study. Pediatr Rheumatol Online J 2020; 18:9. [PMID: 31973755 PMCID: PMC6979350 DOI: 10.1186/s12969-020-0404-8] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/22/2019] [Accepted: 01/06/2020] [Indexed: 01/21/2023] Open
Abstract
BACKGROUNDS In order to provide juvenile idiopathic arthritis (JIA) patients with better pre-conceptional and prenatal counselling, we investigated the obstetrical and neonatal outcomes among women with Asian descent. METHODS Through the linkage of Taiwan National Health Insurance database and National Birth Registry, we established a population-based birth cohort in Taiwan between 2004 and 2014. In a case control study design, first children born to mothers with JIA are identified and matched with 5 non-JIA controls by maternal age and birth year. Conditional logistic regression was used to calculate odds ratios for maternal and neonatal outcomes crude and with adjustment. RESULTS Of the 2,100,143 newborn, 778 (0.037%) were born to JIA mothers. Among them, 549 first-born children were included in this research. Our result suggested that babies born to mothers with JIA were more likely to have low birth body weight, with an adjusted OR of 1.35(95% CI: 1.02 to 1.79) when compared to babies born to mothers without. No differences were observed in other perinatal complications between women with and without JIA including stillbirth, prematurity, or small for gestational age. The rate of adverse obstetrical outcomes such as caesarean delivery, preeclampsia, gestational diabetes, postpartum hemorrhage and mortality were also similar between the two. CONCLUSIONS Adverse obstetrical and neonatal outcomes were limited among Asian mothers with JIA. Intensive care may not be necessary for JIA mothers and their newborns.
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Affiliation(s)
| | - Huang-Yu Yang
- grid.145695.aChang Gung University, College of Medicine, Taoyuan, Taiwan ,0000 0001 0711 0593grid.413801.fDepartment of Nephrology, Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Meng-Jun Chiu
- 0000 0001 0711 0593grid.413801.fCenter for Artificial Intelligence in Medicine, Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - I-Jun Chou
- grid.145695.aChang Gung University, College of Medicine, Taoyuan, Taiwan ,0000 0001 0711 0593grid.413801.fDivision of Neurology, Department of Pediatrics, Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Chang-Fu Kuo
- grid.145695.aChang Gung University, College of Medicine, Taoyuan, Taiwan ,0000 0001 0711 0593grid.413801.fDepartment of Rheumatology, Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Jing-Long Huang
- grid.145695.aChang Gung University, College of Medicine, Taoyuan, Taiwan ,0000 0001 0711 0593grid.413801.fDivision of Allergy, Asthma, and Rheumatology, Department of Pediatrics, Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Kuo-Wei Yeh
- grid.145695.aChang Gung University, College of Medicine, Taoyuan, Taiwan ,0000 0001 0711 0593grid.413801.fDivision of Allergy, Asthma, and Rheumatology, Department of Pediatrics, Chang Gung Memorial Hospital, Taoyuan, Taiwan
| | - Chao-Yi Wu
- Chang Gung University, College of Medicine, Taoyuan, Taiwan. .,Division of Allergy, Asthma, and Rheumatology, Department of Pediatrics, Chang Gung Memorial Hospital, Taoyuan, Taiwan.
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Miyamae T, Tani Y, Kishi T, Yamanaka H, Singh G. Updated version of Japanese Childhood Health Assessment Questionnaire (CHAQ). Mod Rheumatol 2019; 30:905-909. [PMID: 31441680 DOI: 10.1080/14397595.2019.1660027] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/26/2022]
Abstract
Objectives: The Childhood Health Assessment Questionnaire (CHAQ) is one of the most widely used self- report questionnaires to measure functional status in Juvenile idiopathic arthritis (JIA). The Japanese version of the CHAQ (JCHAQ) has been revised to meet requirements of clinical international trials which need the same number of questions in each functional area of the CHAQ.Methods: The original JCHAQ consisted of 36 items, measuring eight functional areas. This was changed to 30 items of questionnaire so that each functional area has same number of questions as the original US English version. The revised version was professionally translated from English to Japanese, reviewed, and validated with Japanese JIA patients.Results: A total of 42 JIA patients were enrolled in the validation: seven systemic, 30 polyarticular/oligoarticular and five enthesis related. Most patients were well controlled and the median disability index (DI) scores was 0.0 [0-0.03]; however, significant correlation was seen with visual analog scale (VAS) of pain, VAS overall well-being, physician VAS, DAS (Disease Activity Score) 28-ESR, and JADAS (Juvenile Arthritis Disease Activity Score)-27. In comparison of two groups of disease activity, remission or inactive/low disease activity vs. moderate/high disease activity, both DAS28-ESR and JADAS-27 showed significant correlation with DI.Conclusion: The updated JCHAQ was a reliable and valid tool for the functional assessment of children with JIA. It is more suitable for international and transitional comparison.
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Affiliation(s)
- Takako Miyamae
- Institute of Rheumatology, Tokyo Women's Medical University, Tokyo, Japan.,Department of Pediatrics, Tokyo Women's Medical University, Tokyo, Japan
| | - Yumi Tani
- Institute of Rheumatology, Tokyo Women's Medical University, Tokyo, Japan.,Department of Pediatrics, Tokyo Women's Medical University, Tokyo, Japan
| | - Takayuki Kishi
- Institute of Rheumatology, Tokyo Women's Medical University, Tokyo, Japan.,Department of Pediatrics, Tokyo Women's Medical University, Tokyo, Japan
| | - Hisashi Yamanaka
- Institute of Rheumatology, Tokyo Women's Medical University, Tokyo, Japan
| | - Gurkirpal Singh
- Division of Gastroenterology and Hepatology, Stanford University, Stanford, CA, USA.,Institute of Clinical Outcomes Research and Education (ICORE), Woodside, CA, USA
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Kısaarslan AP, Sözeri B, Gündüz Z, Zararsız G, Poyrazoğlu H, Düşünsel R. Evaluation of factors affecting the duration of disease-modifying anti-rheumatic drugs application in patients with enthesitis-related arthritis. Eur J Rheumatol 2019; 6:130-135. [PMID: 31070580 DOI: 10.5152/eurjrheum.2019.18180] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/14/2018] [Accepted: 03/05/2019] [Indexed: 11/22/2022] Open
Abstract
OBJECTIVE Treatments for enthesitis-related arthritis (ERA) consist of a mono- or combination therapy with non-steroidal anti-inflammatory drugs, disease-modifying anti-rheumatic drugs (DMARDs), and biological agents, and they are primarily based on adult studies and studies on other forms of juvenile idiopathic arthritis, depending on whether there is axial or peripheral involvement. We use DMARDs frequently in our daily practice, even in patients with axial involvement. The main reason for this is that the health insurance system in Turkey does not allow the use of Tumor Negrosis Factor (TNF) blockers as the first line of treatment. The aim of this study is to evaluate the factors affecting the duration of DMARDs application in patients with ERA. METHODS Fifty-two patients with ERA were accepted in this retrospective cohort study. These patients did not have an inflammatory bowel disease, reactive arthritis or undifferentiated arthritis, psoriasis, and familial Mediterranean fever. Demographic characteristics, medical history, the initial and follow-up physical examination, initial Juvenile Spondyloarthritis Disease Activity Index (JSpADA), initial laboratory tests, radiographic tests, Juvenile Arthritis Damage Index-articulary (JADI-A) and extra-articulary (JADI-E) on the last admission, and data on medical treatments were recorded from the registered data. The univariate Cox proportional hazards regression analyses was used to determine factors affecting the non-response time of ERA patients to DMARDs before the biological treatment was started. RESULTS Twenty-seven patients (52%) achieved remission with DMARDs, while 25 (48%) patients did not. The age at diagnosis (HR=1.12; p=0.247); gender (HR=2.53; p=0.210); family history of ankylosing spondylitis (HR=1.17; p=0.730); inflammatory back pain (HR=0.57; p=0.175); the shoulder (HR=0.75 p=0.706), hip (HR=0.45; p=0.129), and small-joint involvement (HR=1.53; p=0.439); sacroiliitis with physical examination (HR=0.90; p=0.814) and magnetic resonance imaging (MRI) (HR=2.84; p=0.110); enthesitis (HR=0.83; p=0.670); presence of uveitis (HR=2.04; p=0.342); presence of HLA-B27 (HR=1.39; p=0.524); initial high acute phase reactants levels(HR=1.89; p=0.183); initial JSpADA score (HR=0.98; p=0.944); and last JADI-A (HR=1.41; p=0.060) score did not affect the duration of DMARDs treatment before switching to biological treatments. CONCLUSION In our study, the absence of factors affecting the duration of DMARDs application in patients with ERA showed that DMARDs may still be applied as the first line of treatment.
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Affiliation(s)
- Ayşenur Paç Kısaarslan
- Department of Pediatric Rheumatology, Erciyes University School of Medicine, Kayseri, Turkey
| | - Betül Sözeri
- Department of Pediatric Rheumatology, Erciyes University School of Medicine, Kayseri, Turkey
| | - Zübeyde Gündüz
- Department of Pediatric Rheumatology, Erciyes University School of Medicine, Kayseri, Turkey
| | - Gökmen Zararsız
- Department of Biostatistics, Erciyes University School of Medicine, Kayseri, Turkey
| | - Hakan Poyrazoğlu
- Department of Pediatric Rheumatology, Erciyes University School of Medicine, Kayseri, Turkey
| | - Ruhan Düşünsel
- Department of Pediatric Rheumatology, Erciyes University School of Medicine, Kayseri, Turkey
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Nakano N, Mori M, Umebayashi H, Iwata N, Kobayashi N, Masunaga K, Imagawa T, Murata T, Kinjo N, Nagai K, Miyoshi M, Takei S, Yokota S, Ishii E. Characteristics and outcome of intractable vasculitis syndrome in children: Nation-wide survey in Japan. Mod Rheumatol 2017; 28:697-702. [PMID: 29219653 DOI: 10.1080/14397595.2017.1404700] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/18/2022]
Abstract
OBJECTIVE Primary systemic vasculitis (PSV) is a rare disorder in children and difficult to distinguish from other diseases. However, appropriate diagnosis and prompt treatment will affect on the morbidity and mortality of intractable PSV. In this study, we conducted a nationwide survey in Japan, to clarify epidemiology and clinical outcome of PSV. METHODS We had sent survey questionnaires to most of the Japanese institutions that employed pediatricians, requesting the number of patients with refractory PSV who were diagnosed and treated between 2007 and 2011. Respondents were asked to provide detailed information on the clinical and laboratory features of each case they had managed. Those with Kawasaki disease or Henoch-Shönlein purpura vasculitis (IgA vasculitis) were excluded. RESULTS Of all the institutions surveyed, 1123 (37.3%) patients responded, finally, total of 49 patients with intractable PSV, defined by those with resistant to treatment and steroid-dependent, or with any complication associated with prognosis, were selected. The diagnosis was Takayasu arteritis in 31, polyarteritis nodosa in 11, granulomatosis with polyangitis in 2, microscopic polyangitis in 1, and ANCA negative microscopic polyangitis in 1. In those with Takayasu arteritis, 67% were treated with an immunosuppressive agent, 22% with biological modifiers, and 16% with surgical procedures. In other types of disease, 88% of the patients were treated with an immunosuppressive agent, and 12% with biological modifiers. Two with Takayasu arteritis died being terminally ill. CONCLUSION This nationwide survey establishes the heterogeneous characteristics of PSV in children. Although questionnaire-based, the results of our analysis should be useful in planning prospective studies to identify the most effective therapy for each subtype of multifaceted disease.
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Affiliation(s)
- Naoko Nakano
- a Department of Pediatrics , Ehime University Graduate School of Medicine , Ehime , Japan
| | - Masaaki Mori
- b Department of Lifetime Clinical Immunology, Graduate School of Medical and Dental Sciences , Tokyo Medical and Dental University , Tokyo , Japan
| | - Hiroaki Umebayashi
- c Department of General Pediatrics , Miyagi Children's Hospital , Miyagi , Japan
| | - Naomi Iwata
- d Department of Immunology and Infectious Diseases , Aichi Children's Health and Medical Center , Aichi , Japan
| | - Norimoto Kobayashi
- e Department of Pediatrics , Shinshu University School of Medicine , Nagano , Japan
| | - Kenji Masunaga
- f Department of Pediatrics , Kurume University School of Medicine , Fukuoka , Japan
| | - Tomoyuki Imagawa
- g Division of Infection, Immunology and Rheumatology , Kanagawa Children's Medical Center , Kanagawa , Japan
| | - Takuji Murata
- h Department of Pediatrics , Osaka Medical College , Osaka , Japan
| | - Noriko Kinjo
- i Department of Pediatrics , University of the Ryukyu , Okinawa , Japan
| | - Kazushige Nagai
- j Department of Pediatrics , Sapporo Medical University School of Medicine , Hokkaido , Japan
| | - Mari Miyoshi
- k Department of Allergy and Immunology , Hyogo Prefectural Kobe Children's Hospital , Hyogo , Japan
| | - Syuji Takei
- l School of Health Science, Faculty of Medicine , Kagoshima University , Kagoshima , Japan
| | - Shumpei Yokota
- m Department of Pediatrics , Yokohama City University , Kanagawa , Japan
| | - Eiichi Ishii
- a Department of Pediatrics , Ehime University Graduate School of Medicine , Ehime , Japan
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Goyal A, Shah I. Aortoarteritis with tuberculosis. J Family Med Prim Care 2017; 6:153-154. [PMID: 29026771 PMCID: PMC5629883 DOI: 10.4103/2249-4863.214978] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/23/2022] Open
Abstract
Aortoarteritis is an inflammatory condition of the aorta, which has been rarely reported due to tuberculous infection. We report two cases of children who had aortoarteritis along with tuberculosis (TB), of which one had collapse consolidation and the other had latent TB. Both patients were treated with anti-TB therapy and steroids.
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Affiliation(s)
- Anmol Goyal
- Department of Pediatrics, Pediatric TB Clinic, B.J. Wadia Hospital for Children, Mumbai, Maharashtra, India
| | - Ira Shah
- Department of Pediatrics, Pediatric TB Clinic, B.J. Wadia Hospital for Children, Mumbai, Maharashtra, India
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12
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Skeik N, Ostertag-Hill CA, Garberich RF, Alden PB, Alexander JQ, Cragg AH, Manunga JM, Stephenson EJ, Titus JM, Sullivan TM. Diagnosis, Management, and Outcome of Aortitis at a Single Center. Vasc Endovascular Surg 2017; 51:470-479. [PMID: 28859604 DOI: 10.1177/1538574417704296] [Citation(s) in RCA: 21] [Impact Index Per Article: 2.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/24/2022]
Abstract
BACKGROUND Aortitis is a rare condition with inflammatory or infectious etiology that can be difficult to diagnose due to the highly variable clinical presentation and nonspecific symptoms. However, current literature on the diagnosis, management, and prognosis of aortitis is extremely scarce. METHODS We retrospectively reviewed all patients' charts who were diagnosed with giant cell arteritis, Takayasu arteritis, or noninfectious aortitis presenting at a single center between January 1, 2009, and April 17, 2015. Data collected included demographics, medical history, comorbidities, laboratory and imaging data, management, and outcome. RESULTS Among the included 15 patients presenting with aortitis at our center, 53% were diagnosed with Takayasu arteritis, 33% with idiopathic inflammatory aortitis, and 13% with giant cell arteritis. All patients received steroid treatment, 67% received adjunctive immunosuppressants or immunomodulators, and 33% underwent interventional procedures. Based on clinical presentation and laboratory and imaging findings at the last follow-up visit for each patient, 67% showed improvement, 27% had no change in disease activity, and 7% had a progression of the disease. CONCLUSIONS Takayasu arteritis was found to be more common than idiopathic inflammatory aortitis and giant cell arteritis among our 15 cases diagnosed with aortitis. All patients received medical therapy and 33% received interventional procedures, leading to 67% improvement of disease activity or related complications. This article also offers a comprehensive review of the diagnosis, management, and outcome of aortitis, supplementing the very limited literature on this disease.
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Affiliation(s)
- Nedaa Skeik
- 1 Vascular Medicine, Minneapolis Heart Institute, Minneapolis, MN, USA
| | | | - Ross F Garberich
- 2 Minneapolis Heart Institute Research Foundation, Minneapolis, MN, USA
| | - Peter B Alden
- 3 Vascular Surgery, Minneapolis Heart Institute, Minneapolis, MN, USA
| | - Jason Q Alexander
- 3 Vascular Surgery, Minneapolis Heart Institute, Minneapolis, MN, USA
| | - Andrew H Cragg
- 3 Vascular Surgery, Minneapolis Heart Institute, Minneapolis, MN, USA
| | - Jesse M Manunga
- 3 Vascular Surgery, Minneapolis Heart Institute, Minneapolis, MN, USA
| | | | - Jessica M Titus
- 3 Vascular Surgery, Minneapolis Heart Institute, Minneapolis, MN, USA
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13
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Miyamae T, Takei S, Itoh Y, Yamanaka H. Survey of attitudes of non-pediatric rheumatologists among councilors of the Japan College of Rheumatology regarding transitional care. Mod Rheumatol 2017; 27:1047-1050. [DOI: 10.1080/14397595.2017.1285846] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/07/2023]
Affiliation(s)
- Takako Miyamae
- Institute of Rheumatology, Tokyo Women’s Medical University, Tokyo, Japan
- Department of Pediatrics, Tokyo Women’s Medical University, Tokyo, Japan
| | - Syuji Takei
- Kagoshima University Faculty of Medicine School of Health Sciences, Kagoshima, Japan
| | - Yasuhiko Itoh
- Department of Pediatrics, Nippon Medical School, Tokyo, Japan
| | - Hisashi Yamanaka
- Institute of Rheumatology, Tokyo Women’s Medical University, Tokyo, Japan
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14
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Dahman HAB. Challenges in the diagnosis and management of Pediatric Rheumatology in the developing world: Lessons from a newly established clinic in Yemen. Sudan J Paediatr 2017; 17:21-29. [PMID: 29545661 DOI: 10.24911/sjp.2017.2.2] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/11/2022]
Abstract
Pediatric rheumatology is still considered a mysterious branch of pediatric medicine, especially among developing countries. Long-term consequences usually follow delayed diagnosis, referral, and management of rheumatic disorders. We aim to describe the clinical spectrum and the frequency of pediatric rheumatic diseases (PRDs) in AL-Mukalla hospital in Hadhramout province/Yemen. A case record retrospective study was conducted among all patients who attended the pediatric rheumatology clinic in Al-Mukalla hospital (from January 2010 to December 2016) with a musculoskeletal complaint or systemic symptoms suggestive of rheumatic disease. Data collected included: gender, address, age at the onset of symptoms, initial manifestations, the presence of complications, referral diagnosis, final diagnosis and the lag period before attending the rheumatology clinic. PRDs were present in 86% (37/43) of cases, 83.8% (31/37) were from Hadhramout province. Male to female ratio was 0.9:1 (Male: 18; Female: 19) and the commonest age group affected was (>9-12) years. The mean age at first presentation was 8.859 ± 4.11 years (four months-14 years). One year (0.13-98.4 months) was the median time before referral to the rheumatologist. About 70.3% of cases were referred from other specialists or peripheral hospitals. Joint swelling (54.1%) was the most common presentation and juvenile idiopathic arthritis (24.3%) was the commonest diagnosis. The discrepancy between the referral and the final diagnosis was noticed in 21(48.8%) cases. Only eight cases (18.6%) matched the final diagnosis. Two patients died with a mortality rate of 5.4%. Knowledge of the spectrum and incidence of PRDs will increase the awareness of specialists and general practitioners for early referral and diagnosis to avoid long-term sequels.
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Affiliation(s)
- Haifa A Bin Dahman
- Pediatric Department, Hadharmout University College of Medicine (HUCOM), Hadhramout Province, Yemen
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15
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Lin S, Wang Y, Mu S, Zhang J, Yuan F, Sun K. Pathway analysis based on Monte Carlo Cross-Validation in polyarticular juvenile idiopathic arthritis. Pathol Res Pract 2016; 213:7-12. [PMID: 27894617 DOI: 10.1016/j.prp.2016.04.010] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/02/2016] [Accepted: 04/28/2016] [Indexed: 11/18/2022]
Abstract
INTRODUCTION Juvenile idiopathic arthritis (JIA) is a common chronic disease with onset before the 16 years old in a child. Polyarticular JIA has been reported as the main form of JIA in several locations. Until now, understanding of the genetic basis of JIA is incomplete. The purpose of this study was to identify pathway pairs of great potential functional relevance in the progression of polyarticular JIA. MATERIALS AND METHODS Microarray data of 59 peripheral blood samples from healthy children and 61 samples from polyarticular JIA were transformed to gene expression data. Differential expressed genes (DEG) between patients and normal controls were identified using Linear Models for Microarray Analysis. After performed enrichment of DEG, differential pathways were identified with Fisher's test and false discovery rate. Differential pathway pairs were constructed with random two differential pathways, and were evaluated by Random Forest classification. Monte Carlo Cross-Validation was introduced to screen the best pathway pair. RESULTS 42 DEG with P-values<0.01 were identified. 19 differential pathways with P-values<0.01 were identified. Area under the curve (AUC) of pathway pairs was generated with RF classification. After 50 bootstraps of Monte Carlo Cross-Validation, the best pathway pair with the highest AUC value was identified, and it was the pair of tumoricidal function of hepatic natural killer cells pathway and erythropoietin signaling pathway. CONCLUSION These identified pathway pairs may play pivotal roles in the progress of polyarticular JIA and can be applied for diagnosis. Particular attention can be focused on them for further research.
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Affiliation(s)
- Shunhua Lin
- Department of Orthopaedics, The People's Hopital of Rizhao, Rizhao 276800, Shandong, PR China
| | - Yuanji Wang
- Department of Orthopaedics, The People's Hopital of Rizhao, Rizhao 276800, Shandong, PR China
| | - Shunmei Mu
- Department of Ophthalmology, The People's Hopital of Donggang District, Rizhao 276800, Shandong, PR China
| | - Junxi Zhang
- Department of Orthopaedics, The People's Hopital of Rizhao, Rizhao 276800, Shandong, PR China
| | - Fangchang Yuan
- Department of Orthopaedics, The People's Hopital of Rizhao, Rizhao 276800, Shandong, PR China
| | - Kang Sun
- Department of Orthopaedics, The Affiliated Hopital of Qingdao University, Qingdao 266000, Shandong, PR China.
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Lin CH, Lin CL, Shen TC, Wei CC. Epidemiology and risk of juvenile idiopathic arthritis among children with allergic diseases: a nationwide population-based study. Pediatr Rheumatol Online J 2016; 14:15. [PMID: 26965056 PMCID: PMC4787040 DOI: 10.1186/s12969-016-0074-8] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/29/2015] [Accepted: 03/04/2016] [Indexed: 01/19/2023] Open
Abstract
BACKGROUND Epidemiological research reveals that the incidence of allergic diseases and that of autoimmune diseases have been increasing in parallel, raising an interest in a potential link between the two disorders. However, the relationship between Th2-mediated allergic disease and Th1-mediated juvenile idiopathic arthritis (JIA) remains unclear. This population-based case-control study was aimed at investigating the development of childhood-onset allergic diseases and the subsequent risks of JIA. METHODS We included 329 children with JIA diagnosed between 2000 and 2008, and 1316 age- and sex-matched controls. The odds ratios of developing JIA were calculated to determine an association with preexisting allergic diseases. RESULTS The incidence rate of JIA in Taiwan between 2000 and 2008 was 1.33 cases per 100,000 children/year according to the International League of Associations for Rheumatology (ILAR) criteria. The children with a single allergic disease had an increased risk of JIA, with adjusted odds ratios of developing JIA of 1.44 for allergic conjunctivitis (95 % confidence interval [CI], 1.07-1.95), 1.50 for allergic rhinitis (1.15-1.96), and 1.44 for asthma (1.00-2.10). The adjusted odds ratios increased with the number of concurrent allergic diseases from 1.50 (95 % CI, 1.12-2.01) for those with only one allergic disease to 1.72 (1.24-2.38) for those with at least two allergic diseases. The adjusted odds ratios of those with at least two allergic diseases increased to 1.84 (95 % CI, 1.19-2.86) for boys and 2.54 (1.42-4.54) for those older than 12 years. The children who made two or more medical visits for associated allergic diseases per year had an increased risk of JIA. CONCLUSION Children with onset of allergic diseases were at increased risk of developing JIA. The increased risk was associated with the cumulative effect of concurrent allergic diseases and frequency of seeking medical care. Further study to investigate the role of Th2-mediated allergic diseases that contribute to the development of Th1-mediated JIA is warranted.
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Affiliation(s)
- Chien-Heng Lin
| | - Cheng-Li Lin
| | - Te-Chun Shen
| | - Chang-Ching Wei
- Children's Hospital of China Medical University Hospital, Taichung, Taiwan. .,School of Medicine, China Medical University, No2, Yu-Der Road, Taichung, 40402, Taiwan.
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Bossone E, Pluchinotta FR, Andreas M, Blanc P, Citro R, Limongelli G, Della Corte A, Parikh A, Frigiola A, Lerakis S, Ehrlich M, Aboyans V. Aortitis. Vascul Pharmacol 2015; 80:1-10. [PMID: 26721213 DOI: 10.1016/j.vph.2015.11.084] [Citation(s) in RCA: 29] [Impact Index Per Article: 2.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/05/2015] [Revised: 11/22/2015] [Accepted: 11/23/2015] [Indexed: 01/13/2023]
Abstract
Aortitis encompasses all conditions, infectious or non-infectious, leading to different types of inflammation of the aortic wall. The clinical manifestations are usually nonspecific, as are laboratory findings, and sometimes may mimic other diseases. Imaging modalities can help in the assessment of these patients and are often required for making the final diagnosis. It is extremely important to establish an early diagnosis in order to treat timely potentially life-threatening conditions. Anti-inflammatory drugs, antibiotics, sometimes in combination with surgical or endovascular therapies, are proposed according to etiology and disease severity. This review focuses on the classification, epidemiology, diagnostic approach, and management of aortitis with which physicians should be familiar.
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Affiliation(s)
| | - Francesca R Pluchinotta
- Department of Pediatric Cardiology and Adult with Congenital Heart Disease, IRCCS Policlinico San Donato, Milan, Italy
| | - Martin Andreas
- Department of Cardiac Surgery, Medical University of Vienna, Vienna, Austria
| | - Philippe Blanc
- Department of Nuclear Medicine, Dupuytren University Hospital, Limoges, France
| | - Rodolfo Citro
- Heart Department, University Hospital, Salerno, Italy
| | - Giuseppe Limongelli
- Division of Cardiology, Monaldi Hospital, Second University of Naples, Naples, Italy
| | - Alessandro Della Corte
- Department of Cardiothoracic Sciences, Monaldi Hospital, Second University of Naples, Naples, Italy
| | - Ankit Parikh
- Division of Cardiology, Department of Medicine, Emory University School of Medicine, Atlanta, GA, USA
| | - Alessandro Frigiola
- Department of Pediatric Cardiology and Adult with Congenital Heart Disease, IRCCS Policlinico San Donato, Milan, Italy
| | - Stamatios Lerakis
- Division of Cardiology, Department of Medicine, Emory University School of Medicine, Atlanta, GA, USA
| | - Marek Ehrlich
- Department of Cardiac Surgery, Medical University of Vienna, Vienna, Austria
| | - Victor Aboyans
- Department of Cardiology, Dupuytren University Hospital, Limoges, France; Inserm 1094, Limoges School of Medicine, Limoges, France
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Efficacy of Adalimumab in a Girl with Refractory Intestinal Behcet's Disease. Case Rep Rheumatol 2015; 2015:716138. [PMID: 26609459 PMCID: PMC4644826 DOI: 10.1155/2015/716138] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/09/2015] [Revised: 06/10/2015] [Accepted: 06/14/2015] [Indexed: 01/21/2023] Open
Abstract
We describe our experience with a juvenile patient who had refractory intestinal Behcet's disease that responded to adalimumab, a fully humanized antibody against soluble TNF-α and its receptor. The patient, a 13-year-old girl, presented with oral aphthous ulcers, vulvar pain, and rashes on the lower extremities. She gradually developed a low-grade fever, abdominal pain, diarrhea, and hematochezia. Lower gastrointestinal endoscopy revealed ulcers in the terminal ileum, consistent with intestinal Behcet's disease. Methylprednisolone pulse therapy was initiated, after which the symptoms transiently improved, but, during the corticosteroid taper, the abdominal pain recurred. The symptoms resolved soon after the administration of adalimumab. Of importance, the dose of corticosteroids was successfully reduced without exacerbation during 8 months of observation. This is the first reported case in which adalimumab was used for pediatric gastrointestinal Behcet's disease. Adalimumab is a good choice for intestinal Behcet's disease refractory to conventional treatment.
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Abdwani R, Abdalla E, Al Abrawi S, Al-Zakwani I. Epidemiology of juvenile idiopathic arthritis in Oman. Pediatr Rheumatol Online J 2015; 13:33. [PMID: 26232056 PMCID: PMC4522065 DOI: 10.1186/s12969-015-0030-z] [Citation(s) in RCA: 23] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/11/2015] [Accepted: 07/16/2015] [Indexed: 01/21/2023] Open
Abstract
BACKGROUND There is a worldwide variation in the prevalence and subtype distribution of juvenile idiopathic arthritis (JIA) which may be affected by ethnicity and genetic factors. The purpose of this study is to determine the prevalence, subtype distribution and characteristic features of JIA among Omani children and to compare our results with other ethnic populations worldwide. METHODS A population-based, multicenter study among pediatric rheumatology clinics in the Sultanate over a 10 year period between 2004-2013. The diagnosis of JIA and identification of JIA subtypes was based on the ILAR 2004 revised criteria. The hospital charts of these patients were retrospectively reviewed and information was collected. All patients were screened for uveitis by an ophthalmologist using slit lamp examination during regular follow up visits. RESULTS The study included a total of 107 cases of JIA in Oman over the study period. Among the 107 patients, 71 % (n = 77) were girls with a female:male ratio of 2.5:1. The mean age of disease onset was 6.85 ± 3.86 years (range 1-13years) while the mean disease duration of 4.8 ± 2.9 years (range 1-11 years). The incidence of JIA was estimated at 2/100,000 with a prevalence of JIA of 20/100,000. The prevalence of JIA in girls was 28/100,000 while the prevalence in boys was 12/100,000. According to disease distribution, the most frequent subtype was polyarticular JIA rheumatoid factor negative (39.2 %) followed by oligoarthritis (31.8 %), systemic (17.8 %), polyarticular JIA rheumatoid factor positive (7.5 %). The unique feature of the Omani cohort is the lack of occurrence of uveitis. CONCLUSIONS This is the first epidemiological JIA study conducted in Oman that highlights unique geographical disease phenotype. Compared to Western counties, there were higher frequency of polyarticular disease and lack of occurrence of uveitis. Further studies are needed to evaluate the implications of genetic, ethnic and environmental differences of disease expression.
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Affiliation(s)
- Reem Abdwani
- Department of Child Health, Sultan Qaboos University Hospital, P O Box 35, Al Khod, Muscat, PC 123, Sultanate of Oman.
| | - Eiman Abdalla
- Department of Child Health, Sultan Qaboos University Hospital, P O Box 35, Al Khod, Muscat, PC 123, Sultanate of Oman.
| | | | - Ibrahim Al-Zakwani
- Department of Pharmacology & Clinical Pharmacy, College of Medicine & Health Sciences, Sultan Qaboos University, Muscat, Oman. .,Gulf Health Research, Muscat, Oman.
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20
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Kawasaki Y, Ohara S, Miyazaki K, Kanno S, Ono A, Suyama K, Sato M, Suzuki S, Maeda R, Nagasawa K, Hosoya M. Incidence and prognosis of systemic lupus erythematosus in a 35 year period in Fukushima, Japan. Pediatr Int 2015; 57:650-5. [PMID: 25615939 DOI: 10.1111/ped.12588] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/18/2014] [Revised: 11/11/2014] [Accepted: 12/26/2014] [Indexed: 11/26/2022]
Abstract
BACKGROUND We examined the epidemiology, clinical manifestations, and prognosis of pediatric systemic lupus erythematosus (SLE) in Fukushima Prefecture, Japan over a 35 year period. METHODS We collected the medical records of 37 patients diagnosed with SLE between 1977 and 2013. These children were divided into two groups. group 1 consisted of 19 patients who were diagnosed between 1977 and 1995, and group 2 consisted of 18 patients diagnosed between 1996 and 2013. The epidemiology, clinical features, and prognosis were retrospectively compared between the two groups. RESULTS The mean number of patients per 100,000 children per year for group 1 and group 2 was 0.33 ± 0.25 and 0.35 ± 0.30, respectively. The duration from onset of symptoms to treatment in group 2 was shorter than that in group 1, but the clinical and laboratory findings at onset did not differ between the two groups. All patients were treated with prednisolone, and 17 patients in group 1 and 18 in group 2 were treated with methylprednisolone pulse therapy. The frequency of cyclophosphamide treatment decreased whereas the frequency of cyclosporine, tacrolimus and mizoribine pulse therapy increased in group 2. SLE disease activity index (SLEDAI) score at the latest follow up in group 2 was lower in group 1. The survival rate was 84% in group 1 and 100% in group 2. CONCLUSION The frequency and severity of SLE in group 1 were similar to those in group 2, and the prognosis of SLE in group 2 was better than that in group 1.
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Affiliation(s)
- Yukihiko Kawasaki
- Department of Pediatrics, Fukushima Medical University School of Medicine, Fukushima, Japan
| | - Shinichiro Ohara
- Department of Pediatrics, Fukushima Medical University School of Medicine, Fukushima, Japan
| | - Kyohei Miyazaki
- Department of Pediatrics, Fukushima Medical University School of Medicine, Fukushima, Japan
| | - Shuto Kanno
- Department of Pediatrics, Fukushima Medical University School of Medicine, Fukushima, Japan
| | - Atsushi Ono
- Department of Pediatrics, Fukushima Medical University School of Medicine, Fukushima, Japan
| | - Kazuhide Suyama
- Department of Pediatrics, Fukushima Medical University School of Medicine, Fukushima, Japan
| | - Masatoki Sato
- Department of Pediatrics, Some General Hospital, Fukushima, Japan
| | - Shigeo Suzuki
- Department of Pediatrics, Ohara General Hospital, Fukushima, Japan
| | - Ryo Maeda
- Department of Pediatrics, Hoshi General Hospital, Fukushima, Japan
| | | | - Mitsuaki Hosoya
- Department of Pediatrics, Fukushima Medical University School of Medicine, Fukushima, Japan
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Mackie FE, Kainer G, Adib N, Boros C, Elliott EJ, Fahy R, Munro J, Murray K, Rosenberg A, Wainstein B, Ziegler JB, Singh-Grewal D. The national incidence and clinical picture of SLE in children in Australia - a report from the Australian Paediatric Surveillance Unit. Lupus 2015; 24:66-73. [PMID: 25288030 DOI: 10.1177/0961203314552118] [Citation(s) in RCA: 21] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/28/2024]
Abstract
OBJECTIVES The objectives of this paper are to prospectively determine the incidence of paediatric systemic lupus erythematosus (pSLE) in Australia as well as describe the demographics, clinical presentation and one-year outcome. STUDY DESIGN Newly diagnosed cases of pSLE were ascertained prospectively from October 2009 to October 2011 through the Australian Paediatric Surveillance Unit (a national monthly surveillance scheme for notification of childhood rare diseases) as well as national subspecialty groups. Questionnaires were sent to notifying physicians at presentation and at one year. RESULTS The annual incidence rate was 0.32 per 10(5) children aged less than 16 years. The incidence was significantly higher in children of Asian or Australian Aboriginal and Torres Strait Islander parents. Approximately one-third of children underwent a renal biopsy at presentation and 7% required dialysis initially although only one child had end-stage kidney disease (ESKD) at one-year follow-up. CONCLUSION The incidence of pSLE in Australia is comparable to that worldwide with a significantly higher incidence seen in children of Asian and Australian Aboriginal and Torres Strait Islander backgrounds. Renal involvement is common but progression to ESKD, at least in the short term, is rare.
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Affiliation(s)
- F E Mackie
- Department of Nephrology, Sydney Children's Hospital and University of NSW, Australia
| | - G Kainer
- Department of Nephrology, Sydney Children's Hospital and University of NSW, Australia
| | - N Adib
- Queensland Paediatric Rheumatology Services, Australia
| | - C Boros
- Discipline of Paediatrics, University of Adelaide and Rheumatology, Women and Children's Health Network, Adelaide, Australia
| | - E J Elliott
- Discipline of Paediatrics and Child Health, Sydney Medical School, University of Sydney, Australia
| | - R Fahy
- Department of Paediatrics, Alice Springs Hospital, Australia
| | - J Munro
- Rheumatology Unit, Royal Children's Hospital, Melbourne, Australia
| | - K Murray
- Princess Margaret Hospital for Children, Perth, Australia
| | - A Rosenberg
- Department of Nephrology, Sydney Children's Hospital and University of NSW, Australia
| | - B Wainstein
- Department of Immunology, Sydney Children's Hospital, Australia
| | - J B Ziegler
- Department of Immunology, Sydney Children's Hospital, Australia
| | - D Singh-Grewal
- Department of Rheumatology, Sydney Children's Hospital Network, Australia
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23
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Kobayashi I, Mori M, Yamaguchi KI, Ito S, Iwata N, Masunaga K, Shimojo N, Ariga T, Okada K, Takei S. Pediatric Rheumatology Association of Japan recommendation for vaccination in pediatric rheumatic diseases. Mod Rheumatol 2014; 25:335-43. [DOI: 10.3109/14397595.2014.969916] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/31/2022]
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Oberle EJ, Harris JG, Verbsky JW. Polyarticular juvenile idiopathic arthritis - epidemiology and management approaches. Clin Epidemiol 2014; 6:379-93. [PMID: 25368531 PMCID: PMC4216020 DOI: 10.2147/clep.s53168] [Citation(s) in RCA: 23] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/18/2022] Open
Abstract
Juvenile idiopathic arthritis (JIA) is a group of disorders characterized by arthritis persisting for at least 6 weeks with onset before the age of 16 years. Within this cluster of conditions, the polyarticular form (involving more than four joints within the first 6 months) is further divided based on the presence of rheumatoid factor. Children with polyarticular JIA pose unique diagnostic and therapeutic challenges compared to children with involvement of fewer joints. Polyarticular JIA patients tend to have a more refractory course and therefore are at increased risk for joint damage, resulting in poorer functional outcomes and decreased quality of life. Although the ability to treat this disorder continues to improve, especially with the advent of biologic agents, there is still much about the epidemiology and pathogenesis of polyarticular JIA that is unknown. The epidemiology of polyarticular JIA varies worldwide with a vast difference in reported cases between different global regions as well as within individual countries. Several genetic risk loci have been identified conferring increased susceptibility to JIA, many within the human leukocyte antigen region. Beyond the genome, environmental factors also seem to contribute to the etiology of polyarticular JIA. This review article will focus on the epidemiology and current treatments of polyarticular JIA and briefly discuss genetic and environmental influences on the pathogenesis of JIA as well as new and emerging therapies.
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Affiliation(s)
- Edward J Oberle
- Department of Pediatrics, Division of Rheumatology, Medical College of Wisconsin, Milwaukee, WI, USA
| | - Julia G Harris
- Department of Pediatrics, Division of Rheumatology, Medical College of Wisconsin, Milwaukee, WI, USA
| | - James W Verbsky
- Department of Pediatrics, Division of Rheumatology, Medical College of Wisconsin, Milwaukee, WI, USA
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Meyer A, Meyer N, Schaeffer M, Gottenberg JE, Geny B, Sibilia J. Incidence and prevalence of inflammatory myopathies: a systematic review. Rheumatology (Oxford) 2014; 54:50-63. [DOI: 10.1093/rheumatology/keu289] [Citation(s) in RCA: 175] [Impact Index Per Article: 15.9] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/12/2022] Open
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26
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Miyamae T, Tanaka E, Kishi T, Matsuyama T, Igarashi T, Fujikawa S, Taniguchi A, Momohara S, Yamanaka H. Long-term outcome of 114 adult JIA patients in a non-pediatric rheumatology institute in Japan. Mod Rheumatol 2014; 25:62-6. [DOI: 10.3109/14397595.2014.929558] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
Affiliation(s)
- Takako Miyamae
- Institute of Rheumatology, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
- Department of Pediatrics, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
| | - Eiichi Tanaka
- Institute of Rheumatology, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
| | - Takayuki Kishi
- Institute of Rheumatology, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
- Department of Pediatrics, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
| | - Tsuyoshi Matsuyama
- Institute of Rheumatology, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
| | - Tohru Igarashi
- Institute of Rheumatology, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
- Department of Pediatrics, Nippon Medical School, Tokyo, Japan
| | | | - Atsuo Taniguchi
- Institute of Rheumatology, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
| | - Shigeki Momohara
- Institute of Rheumatology, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
| | - Hisashi Yamanaka
- Institute of Rheumatology, Tokyo Women's Medical University, Shinjuku-ku, Tokyo, Japan
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Sugimoto K, Miyazawa T, Nishi H, Izu A, Enya T, Okada M, Takemura T. Childhood Cogan syndrome with aortitis and anti-neutrophil cytoplasmic antibody-associated glomerulonephritis. Pediatr Rheumatol Online J 2014; 12:15. [PMID: 24803850 PMCID: PMC4011777 DOI: 10.1186/1546-0096-12-15] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/25/2014] [Accepted: 04/18/2014] [Indexed: 11/26/2022] Open
Abstract
Cogan syndrome is a systemic disease manifesting interstitial keratitis, sensorineural hearing loss, tinnitus, and rotatory vertigo. Renal complications of this syndrome are very rare. We encountered an adolescent with Cogan syndrome complicated by aortitis and anti-neutrophil cytoplasmic antibody (ANCA)-associated glomerulonephritis. At the age of 14, the patient showed proteinuria in a screening urinalysis at school and was found to lack a right radial pulse. Magnetic resonance angiography disclosed right subclavian artery stenosis. Examination of a renal biopsy specimen showed ANCA-positive crescentic glomerulonephritis. Steroid and immunosuppressant treatment improved renal function and histopathology, but repeated recurrences followed. At 18, the patient developed rotatory vertigo, a sense of ear fullness, and sensorineural hearing loss. The patient was diagnosed with Cogan syndrome. We know of no previous description of ANCA-positive crescentic glomerulonephritis in children with Cogan syndrome. Accordingly, evaluation of aortitis in childhood should include not only otolaryngologic and ophthalmologic examinations, but also periodic urine examination and renal function tests.
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Affiliation(s)
- Keisuke Sugimoto
- Department of Pediatrics, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-Sayama 589-8511, Japan
| | - Tomoki Miyazawa
- Department of Pediatrics, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-Sayama 589-8511, Japan
| | - Hitomi Nishi
- Department of Pediatrics, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-Sayama 589-8511, Japan
| | - Akane Izu
- Department of Pediatrics, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-Sayama 589-8511, Japan
| | - Takuji Enya
- Department of Pediatrics, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-Sayama 589-8511, Japan
| | - Mitsuru Okada
- Department of Pediatrics, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-Sayama 589-8511, Japan
| | - Tsukasa Takemura
- Department of Pediatrics, Kinki University Faculty of Medicine, 377-2 Ohno-higashi, Osaka-Sayama 589-8511, Japan
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Kishi T, Miyamae T, Hara R, Nakajima S, Imagawa T, Mori M, Yokota S. Clinical analysis of 50 children with juvenile dermatomyositis. Mod Rheumatol 2014. [DOI: 10.3109/s10165-012-0647-4] [Citation(s) in RCA: 23] [Impact Index Per Article: 2.1] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
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29
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Yokota S, Mori M, Imagawa T, Takei S, Murata T, Tomiita M, Ito Y, Fujikawa S. Proposal for juvenile idiopathic arthritis guidance on diagnosis and treatment for primary care pediatricians and nonpediatric rheumatologists (2007). Mod Rheumatol 2014. [DOI: 10.3109/s10165-007-0625-4] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022]
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Yu HH, Chen PC, Wang LC, Lee JH, Lin YT, Yang YH, Lin CP, Chiang BL. Juvenile idiopathic arthritis-associated uveitis: a nationwide population-based study in Taiwan. PLoS One 2013; 8:e70625. [PMID: 23940609 PMCID: PMC3734244 DOI: 10.1371/journal.pone.0070625] [Citation(s) in RCA: 34] [Impact Index Per Article: 2.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/13/2013] [Accepted: 06/19/2013] [Indexed: 01/22/2023] Open
Abstract
OBJECTIVE The incidence and prevalence of juvenile idiopathic arthritis (JIA) vary widely across the world but data in East Asia is lacking. Uveitis is a serious cause of morbidity in JIA. This study aimed to analyze the incidence and prevalence of JIA, and the characteristics of JIA-associated uveitis in Taiwan. METHODS A population-based cohort study was conducted using the Taiwan National Health Insurance Research Database. Each patient was individually tracked from 1999 to 2009 to identify the diagnosis of JIA and uveitis using the International Classification of Diseases diagnostic codes. Multivariate logistic regression was used to determine the risk factors and complications of uveitis in patients with JIA. RESULTS The study cohort had 2636 cases of JIA and included juvenile rheumatoid arthritis (57.7%), enthesitis-related arthritis (ERA) (39.2%), and psoriatic arthritis (3.1%). The average annual incidence of JIA and JIA-associated uveitis were 4.93 (range, 3.93-6.23) and 0.25 (range, 0.12-0.37) cases per 100,000 population, respectively. The average period prevalence of JIA was 33.8 cases per 100,000 population. Uveitis occurred in 4.7% of patients with JIA, while JIA-associated uveitis was complicated by cataract (11.2%) and glaucoma (24.8%). Enthesitis-related arthritis was significantly associated with uveitis (OR: 3.47; 95% CI: 2.24-5.37) (p<0.0001). Uveitis diagnosed before JIA was the most significant risk factor for complications of glaucoma or cataract (OR: 3.54; 95% CI: 1.44-8.72) (p = 0.006). CONCLUSIONS The incidence of JIA is low but that of JIA-associated uveitis is increasing. Higher percentage of males in patients with ERA and the strong association between ERA and uveitis are unique for children with JIA in Taiwan. Uveitis diagnosed before arthritis is an important risk factor for complications. Continuous ophthalmologic follow-up is needed for children with JIA or uveitis of unknown etiology.
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Affiliation(s)
- Hsin-Hui Yu
- Department of Pediatrics, National Taiwan University Hospital, Taipei, Taiwan
| | - Pau-Chung Chen
- Institute of Occupational Medicine and Industrial Hygiene, National Taiwan University College of Public Health, Taipei, Taiwan
| | - Li-Chieh Wang
- Department of Pediatrics, National Taiwan University Hospital, Taipei, Taiwan
| | - Jyh-Hong Lee
- Department of Pediatrics, National Taiwan University Hospital, Taipei, Taiwan
| | - Yu-Tsan Lin
- Department of Pediatrics, National Taiwan University Hospital, Taipei, Taiwan
| | - Yao-Hsu Yang
- Department of Pediatrics, National Taiwan University Hospital, Taipei, Taiwan
| | - Chang-Ping Lin
- Department of ophthalmology, National Taiwan University Hospital, Taipei, Taiwan
| | - Bor-Luen Chiang
- Department of Medical Research, National Taiwan University Hospital, Taipei, Taiwan
- * E-mail:
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Yeh KW, Yu CH, Chan PC, Horng JT, Huang JL. Burden of systemic lupus erythematosus in Taiwan: a population-based survey. Rheumatol Int 2013; 33:1805-11. [PMID: 23314932 DOI: 10.1007/s00296-012-2643-6] [Citation(s) in RCA: 50] [Impact Index Per Article: 4.2] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/16/2012] [Accepted: 12/15/2012] [Indexed: 12/29/2022]
Abstract
This population-based study aimed to determine the trend of incidence, prevalence, and mortality of systemic lupus erythematosus (SLE) in a 6-year period in Taiwan. Patients with international classification of diseases ninth revision (ICD-9) code 710.0 were retrieved from the Taiwanese National Health Insurance Research Database (NHIRD), which covered more than 96 % of the entire population, and from the Ministry of Interior between 2003 and 2008 in Taiwan. Patients with SLE registered as catastrophic illness were enrolled for analysis. The incidence rate, prevalence ratio, and mortality rate stratified by sex and age were analyzed. There were a total of 6,675 SLE patients (5,836 females and 839 in males) during the study period. The average annual incidence rate was 4.87 per 100,000 population, and the average female-to-male incidence ratio was 7.15. The ratio increased with age and peaked at the age of 40-49 years, then decreased thereafter. The incidence rate decreased by 4.2 % per year. The highest incidence rate was noted in the 20-29-year-old age group in females and the 70-79-year-old age group in males. The average prevalence and mortality rates were 97.5 and 1.2 per 100,000 population, respectively. Mortality was 3.2 % in patients diagnosed within 1 year and is more prevalent in young patients with average age of 15.6 years. Incidence rate of SLE has been declining in recent years but the prevalence rate has remained steady. The highest mortality rate is among younger patients diagnosed with SLE within 1 year.
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Affiliation(s)
- Kuo-Wei Yeh
- Division of Pediatric Allergy Asthma and Rheumatology, Department of Pediatrics, Chang Gung Memorial Hospital at Linkuo, and College of Medicine, Chang Gung University, 5, Fu-Hsin Street, Taoyuan, Taiwan
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Clinical analysis of 50 children with juvenile dermatomyositis. Mod Rheumatol 2012; 23:311-7. [PMID: 22526832 DOI: 10.1007/s10165-012-0647-4] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/12/2012] [Accepted: 04/02/2012] [Indexed: 10/28/2022]
Abstract
OBJECTIVE We performed a retrospective review of medical records to assess the clinical characteristics of 50 Japanese children with juvenile dermatomyositis (JDM). METHODS Fourteen boys and 36 girls who visited Yokohama City University Hospital between 1983 and 2008 were enrolled. Gender, age at disease onset and diagnosis, presenting clinical features, laboratory data at onset, complications, treatment, and outcome were reviewed. RESULTS Mean age at disease onset was 6.9 years. Clinical manifestations at the first visit were muscle pain and/or weakness (90 %), malar rash (90 %), Gottron's papules (86 %), and heliotrope rash (80.0 %). Elevated serum levels of creatine kinase were found in 57.0 % of patients and aldolase in 95 %. T2-weighted magnetic resonance (MR) images with fat suppression demonstrated positive findings in 89.5 % of patients. Initial treatment was prednisolone (PSL) orally or pulsed methylprednisolone (mPSL) i.v. Pulsed mPSL therapy showed efficacy superior to PSL [flare in 8 of 19 (42 %) vs. 18 of 25 (72 %)]. Children refractory to initial treatment were given additional pulsed mPSL and/or cyclophosphamide (IVCY; n = 19) i.v.. Four patients with interstitial pneumonia responded well to IVCY. CONCLUSIONS Our findings support the notion that JDM might be considered as both a systemic inflammatory and noninflammatory vasculopathy best treated by IVCY, as shown in previous literature.
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Abstract
Juvenile idiopathic arthritis (JIA) encompasses a complex group of disorders with arthritis as a common feature. This article provides the pediatrician with a review of the epidemiology, classification, clinical manifestations, and complications of JIA. It also provides an update on the current understanding of the cause of JIA and recent developments in management and a recent review of the long-term outcome in JIA.
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Affiliation(s)
- Peter J Gowdie
- Division of Rheumatology, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada
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Takahashi K, Oharaseki T, Yokouchi Y, Yamada H, Shibuya K, Naoe S. A half-century of autopsy results--incidence of pediatric vasculitis syndromes, especially Kawasaki disease. Circ J 2012; 76:964-70. [PMID: 22313802 DOI: 10.1253/circj.cj-11-0928] [Citation(s) in RCA: 21] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/09/2022]
Abstract
BACKGROUND The objectives of this study were to clarify the details of pediatric vasculitic diseases on the basis of Japanese autopsy reports and determine whether there were cases of probable Kawasaki disease (KD) even before KD came to be widely recognized as a disease entity. METHODS AND RESULTS Systemic vasculitis autopsy cases aged 15 years or less were selected from the total of 1,335,045 autopsy cases listed in the Annual of Pathological Autopsy Cases in Japan from 1958 through 2008. Those cases were classified into 14 disease groups and then analyzed with regard to various details. There were 380 autopsy cases of vasculitis in children (0.03% of the total autopsy cases). More than half were KD, and other diseases included unclassified vasculitis, polyarteritis nodosa, purpuric vasculitis, Takayasu arteritis, etc. The first recorded case of KD autopsy occurred in 1969. Up until 1976 there was a great difference in the number of autopsies between pediatric vasculitis and KD. However, after 1977 their numbers were in close agreement. The autopsy findings for 24 of 125 child vasculitis autopsies performed before 1976 and diagnosed as non-KD were consistent with KD. CONCLUSIONS Although autopsies of pediatric vasculitis cases are extremely rare, the majority consists of KD. Moreover, it is likely that autopsy cases that were probably KD first appeared in the early 1960s.
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Affiliation(s)
- Kei Takahashi
- Department of Pathology, Toho University Ohashi Medical Center, Tokyo 153-8515, Japan.
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Reiff A. Treatment of Systemic Juvenile Idiopathic Arthritis with Tocilizumab - the Role of Anti-Interleukin-6 Therapy After a Decade of Treatment. BIOLOGICS IN THERAPY 2012; 2:1. [PMID: 24392296 PMCID: PMC3873121 DOI: 10.1007/s13554-012-0001-6] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 12/08/2011] [Indexed: 12/17/2022]
Abstract
With the recent approval of tocilizumab as the first biologic for the treatment of systemic juvenile idiopathic arthritis (sJIA), an important unmet medical need for this historically challenging disease has now been met. The purpose of this review article is to revisit the established therapeutic options for sJIA, to summarize the history of the clinical trials with tocilizumab, and to discuss its role in the treatment of sJIA.
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Affiliation(s)
- Andreas Reiff
- Division of Rheumatology, Childrens Hospital Los Angeles, Los Angeles, CA USA
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36
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Zhu J, Wu F, Huang X. Age-related differences in the clinical characteristics of systemic lupus erythematosus in children. Rheumatol Int 2012; 33:111-5. [PMID: 22228466 DOI: 10.1007/s00296-011-2354-4] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/13/2011] [Accepted: 12/22/2011] [Indexed: 11/30/2022]
Abstract
The objective of this study was to determine how the clinical presentation of systemic lupus erythematosus in pediatric varied with the age of onset of the disease. We reviewed the charts of a total of 88 Chinese pediatric patients (pSLE) diagnosed and admitted first time to our hospitals between 2005 and 2008. Patients were divided into 3 groups, depending on the age at diagnosis: preschool (1-6 years), school age (7-11 years), and adolescent groups (12-18 years). Among the three groups, we compared the sex ratio, disease duration at diagnosis, symptoms at the onset of the disease, clinical manifestations, laboratory examinations, SLEDAI (Systemic Lupus Erythematosus Disease Activity Index) 2000, and SLICC/ACR SDI (Systemic Lupus International Collaborating Clinics/American College of Rheumatology Damage Index for Systemic Lupus Erythematosus). Most pediatric patients were in the adolescent group (47.7%), while the disease duration at diagnosis was shortest in preschool-age patients (0.65 ± 0.5 months). The most common symptoms at the onset of pSLE were fever, rash, arthritis, and seizures. Hematologic system and neuropsychiatric system were damaged commonly. The preschool-age patients had the shortest disease duration at diagnosis, the highest incidence of neuropsychiatric system involvement, and the poorest prognosis of all the age groups, especially the boys. Patients in adolescence had the peak incidence of pediatric SLE and high disease activity, begins to acquire some of the adult characteristics. School-age is a transition stage between other stages.
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Affiliation(s)
- Jia Zhu
- Division of Pediatric Rheumatology, Children's Hospital Affiliated Capital Institute of Pediatrics, No.2 Yabao Road, Chaoyang District, Beijing 100020, China
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Pineles D, Valente A, Warren B, Peterson MGE, Lehman TJA, Moorthy LN. Worldwide incidence and prevalence of pediatric onset systemic lupus erythematosus. Lupus 2011; 20:1187-92. [DOI: 10.1177/0961203311412096] [Citation(s) in RCA: 71] [Impact Index Per Article: 5.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Abstract
Compilation of worldwide data regarding the incidence and prevalence of pediatric-onset systemic lupus erythematosus (SLE) is needed in order to evaluate the scope of the disease in the pediatric population. A literature review was performed to unify the current data available on the global incidence and prevalence of pediatric-onset SLE. We examined 13 available epidemiological studies concentrated on the incidence and prevalence of pediatric-onset SLE. The available reports were predominantly from North America, Europe and Asia. The limited amount of studies available highlights the need for more epidemiological research in order to better comprehend the global scope of this disease.
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Affiliation(s)
- D Pineles
- Yeshiva University, New York, NY, USA
| | | | | | | | - TJA Lehman
- Hospital for Special Surgery, New York, NY, USA
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Huang JL, Yeh KW, Yao TC, Huang YL, Chung HT, Ou LS, Lee WI, Chen LC. Pediatric lupus in Asia. Lupus 2011; 19:1414-8. [PMID: 20947550 DOI: 10.1177/0961203310374339] [Citation(s) in RCA: 42] [Impact Index Per Article: 3.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/16/2022]
Abstract
Of all patients with systemic lupus erythematosus (SLE), 15-20% are diagnosed during childhood, with disease onset prior to the age of 16 years. Because disease expression in SLE is influenced by environment factors and differs between racial and ethnic groups. The aims of this review were to describe prevalence, clinical manifestations, common infectious complications, and outcome of pediatric-onset SLE in Asia. The prevalence of pediatric-onset SLE was 6.3-19.3 per 100,000 in Asia. The ratio of female to male was 4.7-6.2. The mean age at diagnosis of pediatric-onset SLE was 8.6-13.5 years. The most common clinical features of pediatric-onset SLE in Asia were cutaneous rashes, arthritis, hematological involvement and nephritis. The occurrence of nephritis varies from 29% to 81%. The most common histopathology of lupus nephritis was diffuse proliferative glomerulonephritis (WHO Class-IV) which occurred in 39.4-54% of case of lupus nephritis. Pediatric-onset SLE patients with infections have poor outcomes than uninfected patients. Gram-negative bacilli are the most common microorganisms responsible for bacteremia in Asian patients with SLE. Recurrent major infections predict poorer disease outcome and associated organ damage in pediatric-onset SLE. Improving the survival of SLE patients was reported in Asia in recent decades. The survival was 92% at the age of 5 years, 86% at 10 years and 79% at 15 years in children with SLE in Taiwan in 2008.
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Affiliation(s)
- J L Huang
- Department of Pediatrics, Chang Gung Memorial Hospital, and Chang Gung University, Taoyuan, Taiwan.
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39
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Gulay CB, Dans LF. Clinical presentations and outcomes of Filipino juvenile systemic lupus erythematosus. Pediatr Rheumatol Online J 2011; 9:7. [PMID: 21306603 PMCID: PMC3045886 DOI: 10.1186/1546-0096-9-7] [Citation(s) in RCA: 22] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/11/2010] [Accepted: 02/09/2011] [Indexed: 11/24/2022] Open
Abstract
OBJECTIVE Juvenile Systemic Lupus Erythematosus (SLE) varies by location and ethnicity. This study describes the clinical, laboratory profile and outcome of juvenile SLE seen at Philippine General Hospital (PGH) from 2004-2008. METHOD Medical charts of all Filipino Juvenile SLE cases admitted at PGH during the 5-year period were reviewed collecting demographic profile, clinical and laboratory manifestations and treatment during disease course. RESULTS Seventy-eight cases of juvenile SLE were reviewed. There were 7 boys and 71 girls. The mean age at diagnosis was 14 years (SD 2.7) with a range of 8-18 years. Fever (52.5%) and malar rash (41.0%) were the most common features at disease onset. At the time of diagnosis, the most common features were malar rash (65.3%), renal involvement (62.8%) and photosensitivity (55.1%). Mucocutaneous (92.3%), renal (71.7%) and hematologic (69.2%) involvement were the most common features during the entire course of illness. Infection (34.5%) and neurologic (19.0%) complications were observed most frequently. Corticocosteroid treatment was given in most of the patients in the form of prednisone (97.4%) and concomitant methylprednisolone intravenous pulses (29.4%). Nine patients died during the study period. The overall 5-year mortality rate was 11.5%. Infection (77.0%) was the most frequent cause of death. CONCLUSION Malar rash was a common feature at disease onset and at diagnosis among Filipinos with juvenile SLE. Throughout the disease course, renal involvement occurs in 71.7% of patients. Infection was the leading cause of complication and death. The clinical presentations of Filipinos with juvenile SLE were similar to juvenile SLE in other countries.
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Affiliation(s)
- Carien B Gulay
- Section of Pediatric Rheumatology, Department of Pediatrics, Philippine General Hospital-University of the Philippines Manila, Taft Avenue, Manila, Philippines.
| | - Leonila F Dans
- Section of Pediatric Rheumatology, Department of Pediatrics, Philippine General Hospital-University of the Philippines Manila, Taft Avenue, Manila, Philippines
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40
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Minden K. Classification and epidemiology of juvenile idiopathic arthritis. Rheumatology (Oxford) 2011. [DOI: 10.1016/b978-0-323-06551-1.00097-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/28/2022] Open
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Pediatric Rheumatology in Southeast Asia: Insights from the Singapore Experience. Curr Rheumatol Rep 2010; 13:117-22. [PMID: 21181313 DOI: 10.1007/s11926-010-0159-1] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/18/2022]
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Hahn YS, Kim JG. Pathogenesis and clinical manifestations of juvenile rheumatoid arthritis. KOREAN JOURNAL OF PEDIATRICS 2010; 53:921-30. [PMID: 21218013 PMCID: PMC3012271 DOI: 10.3345/kjp.2010.53.11.921] [Citation(s) in RCA: 30] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 10/04/2010] [Accepted: 10/19/2010] [Indexed: 12/24/2022]
Abstract
Juvenile rheumatoid arthritis (JRA) is the most common rheumatic childhood disease; its onset is before 16 years of age and it persists for at least 6 weeks. JRA encompasses a heterogeneous group of diseases that is classified according to 3 major presentations: oligoarthritis, polyarthritis, and systemic onset diseases. These presentations may originate from the same or different causes that involve interaction with specific immunogenetic predispositions, and result in heterogeneous clinical manifestations. An arthritic joint exhibits cardinal signs of joint inflammation, such as swelling, pain, heat, and loss of function; any joint can be arthritic, but large joints are more frequently affected. Extra-articular manifestations include high fever, skin rash, serositis, and uveitis. The first 2 types of JRA are regarded as T helper 1 (Th1) cell-mediated inflammatory disorders, mainly based on the abundance of activated Th1 cells in the inflamed synovium and the pathogenetic role of proinflammatory cytokines that are mainly produced by Th1 cell-stimulated monocytes. In contrast, the pathogenesis of systemic onset disease differs from that of other types of JRA in several respects, including the lack of association with human leukocyte antigen type and the absence of autoantibodies or autoreactive T cells. Although the precise mechanism that leads to JRA remains unclear, proinflammatory cytokines are thought to be responsible for at least part of the clinical symptoms in all JRA types. The effectiveness of biologic therapy in blocking the action of these cytokines in JRA patients provides strong evidence that they play a fundamental role in JRA inflammation.
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Affiliation(s)
- Youn-Soo Hahn
- Department of Pediatrics, Chungbuk National University College of Medicine, Cheongju, Korea
| | - Joong-Gon Kim
- Department of Pediatrics, Seoul National University College of Medicine, Seoul, Korea
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Ellis JA, Munro JE, Ponsonby AL. Possible environmental determinants of juvenile idiopathic arthritis. Rheumatology (Oxford) 2009; 49:411-25. [DOI: 10.1093/rheumatology/kep383] [Citation(s) in RCA: 67] [Impact Index Per Article: 4.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/20/2023] Open
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Hiraki LT, Benseler SM, Tyrrell PN, Harvey E, Hebert D, Silverman ED. Ethnic differences in pediatric systemic lupus erythematosus. J Rheumatol 2009; 36:2539-46. [PMID: 19833755 DOI: 10.3899/jrheum.081141] [Citation(s) in RCA: 114] [Impact Index Per Article: 7.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/27/2023]
Abstract
OBJECTIVE Prevalence and severity of systemic lupus erythematosus (SLE) in adults is suggested to be distinctly different between ethnic groups. The impact of ethnicity is not as well delineated in pediatric SLE (pSLE). We compared prevalence and extent of major organ involvement, disease activity, and damage in pSLE between different ethnic groups. METHODS Ethnic demographic profiles of an inception cohort of 265 patients with pSLE followed at Sick Kids Hospital in Toronto were determined and compared to the Metropolitan Toronto at-risk population. Patients were categorized into ethnic subsets based on self-designated ethnic origins. Disease characteristics including major organ involvement, disease activity, and damage measures were longitudinally determined and compared among ethnic groups. RESULTS Ethnicity data were available on 259/265 pSLE patients (99.6%); the majority were non-Caucasian (60%) compared to the Metropolitan Toronto at-risk population (40%) (p < 0.0001). Non-Caucasian patients were younger at diagnosis than Caucasian patients, Black patients being the youngest at diagnosis (12.6 vs 14.6 yrs; p = 0.007). Renal disease was significantly more common in non-Caucasian than in Caucasian pSLE patients (62% vs 45%; p = 0.01). There was a trend toward increased prevalence of central nervous system disease in Black patients compared to Asian patients (p = 0.108). There was no difference in gender ratio, SLE Disease Activity Index, or damage scores between ethnic groups. CONCLUSION Non-Caucasian ethnicity is associated with increased pSLE disease prevalence. Non-Caucasian pSLE patients were significantly younger and more likely to have nephritis. However, disease activity and damage were strongly associated with major organ disease independent of the patient's ethnicity.
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Affiliation(s)
- Linda T Hiraki
- Division of Rheumatology, Hospital for Sick Children, University of Toronto, Toronto, Canada
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Abstract
Juvenile idiopathic arthritis (JIA) refers to a group of chronic childhood arthropathies of unknown etiology, currently classified into subtypes primarily on the basis of clinical features. Research has focused on the hypothesis that these subtypes arise through distinct etiologic pathways. In this Review, we discuss four subtypes of JIA: persistent oligoarticular, extended oligoarticular, rheumatoid-factor-positive polyarticular and rheumatoid-factor-negative polyarticular. These subtypes differ in prevalence between ethnic groups and are associated with different HLA alleles. Non-HLA genetic risk factors have also been identified, some of which reveal further molecular differences between these subtypes, while others suggest mechanistic overlap. Investigations of immunophenotypes also provide insights into subtype differences: adaptive immunity seems to have a prominent role in both polyarticular and oligoarticular JIA, and the more-limited arthritis observed in persistent oligoarticular JIA as compared with extended oligoarticular JIA may reflect more-potent immunoregulatory T-cell activity in the former. Tumor necrosis factor seems to be a key mediator of both polyarticular and oligoarticular JIA, especially in the extended oligoarticular subtype, although elevated levels of other cytokines are also observed. Limited data on monocytes, dendritic cells, B cells, natural killer T cells and neutrophils suggest that the contributions of these cells differ across subtypes of JIA. Within each subtype, however, common pathways seem to drive joint damage.
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Methotrexate for the treatment of juvenile idiopathic arthritis: process to approval for JIA indication in Japan. Mod Rheumatol 2008; 19:1-11. [PMID: 18815725 PMCID: PMC2638602 DOI: 10.1007/s10165-008-0123-3] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/22/2008] [Accepted: 08/19/2008] [Indexed: 11/20/2022]
Abstract
Methotrexate (MTX), the primary treatment for the articular-type juvenile idiopathic arthritis (JIA), is effective and brings about radiological improvement. Patient compliance is good, and it is recognized that its known side effects, namely, disruption of liver function and induction of pulmonary lesions, are unlikely to be severe at the low MTX doses that are administered. In Japan, MTX was granted approval in 1999 by the then Ministry of Health and Welfare specifically for treating rheumatoid arthritis in adult patients, allowing it be generally used in medical institutions for patients having National Health Insurance. However, in the pediatric field, its use outside the indications has so far been unavoidable, and has been left to the discretion of the physician. Finally, at the present conference, expansion of the indications of MTX for JIA was approved in Japan. It is noteworthy that this expansion of indications was achieved without requiring clinical trials on children sponsored by the pharmaceutical company: it was achieved rather by collecting necessary information through ongoing efforts (including collection and analysis of information about approval status in foreign countries, adequate evidence from the literature, implementation of a clinical use survey in Japan, etc.). It also merits attention that the maximum dose (10 mg/m2) was set on the basis of pharmacokinetic data from children, rather than relying on the dosing method and dose for adults.
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Affiliation(s)
- Heather L. Gornik
- Assistant Professor of Medicine, Cleveland Clinic Lerner College of Medicine of Case Western Reserve University Medical Director, Non-Invasive Vascular Laboratory Department of Cardiovascular Medicine The Cleveland Clinic Foundation 9500 Euclid Avenue/Desk S60 Cleveland, Ohio 44120 (216) 445-3689
| | - Mark A. Creager
- Professor of Medicine, Harvard Medical School Simon C. Fireman Scholar in Cardiovascular Medicine Director, Vascular Center Brigham and Women's Hospital 75 Francis Street Boston, Massachusetts 02115 (617) 732-5267
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Yokota S, Mori M, Imagawa T, Takei S, Murata T, Tomiita M, Ito Y, Fujikawa S. Proposal for juvenile idiopathic arthritis guidance on diagnosis and treatment for primary care pediatricians and nonpediatric rheumatologists (2007). Mod Rheumatol 2007; 17:353-63. [PMID: 17929125 DOI: 10.1007/s10165-007-0625-4] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/26/2007] [Accepted: 08/29/2007] [Indexed: 10/22/2022]
Abstract
The Pediatric Standing Committee of the Japan College of Rheumatology, in collaboration with the Pediatric Rheumatology Association of Japan, produced guidance on the diagnosis and treatment for juvenile idiopathic arthritis (JIA) for primary care pediatricians and nonpediatric rheumatologists in Japan. This guidance aims to achieve early diagnosis and treatment for JIA, which is similar to adult rheumatoid arthritis (RA), based on recent progress in rheumatology, and to resolve arthritis at an early stage and improve the prognosis of the affected inflammatory joints. It describes clinical symptoms and laboratory findings characteristic to JIA in order to make early diagnosis and treatment possible, and also serves as a triage of patients who are refractory to the treatment protocol described here and need more aggressive interventions. However, because JIA is a complicated and heterogeneous disease and the optimal treatment approach can be diverse and different patient by patient, these guidelines should be viewed as recommendations and be individualized according to the condition of the patient. Finally, we hope that this guidance will trigger exploration for further information by referring to the textbooks and literature listed at the end of these guidelines.
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Affiliation(s)
- Shumpei Yokota
- Department of Pediatrics, Yokohama City University School of Medicine, 3-9 Fukuura, Kanazawa-ku, Yokohama, 236-0004, Japan.
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Saurenmann RK, Rose JB, Tyrrell P, Feldman BM, Laxer RM, Schneider R, Silverman ED. Epidemiology of juvenile idiopathic arthritis in a multiethnic cohort: ethnicity as a risk factor. ACTA ACUST UNITED AC 2007; 56:1974-84. [PMID: 17530723 DOI: 10.1002/art.22709] [Citation(s) in RCA: 181] [Impact Index Per Article: 10.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/06/2023]
Abstract
OBJECTIVE To study the influence of ethnicity on the risk of developing juvenile idiopathic arthritis (JIA) in a multiethnic community of patients with unrestricted access to health care. METHODS A questionnaire on ethnicity was distributed to all patients with JIA being followed up at the Hospital for Sick Children in Toronto, Ontario, Canada. Of 1,082 patients, 859 (79.4%) responded to the questionnaire. To calculate the relative risk (RR) of developing JIA in this study cohort, the results were compared with data from the age-matched general population of the Toronto metropolitan area (TMA) as provided in the 2001 census from Statistics Canada. RESULTS European descent was reported by 69.7% of the patients with JIA compared with a frequency of 54.7% in the TMA general population, whereas a statistically significantly lower than expected percentage of the patients with JIA reported having black, Asian, or Indian subcontinent origin. Children of European origin had a higher RR for developing any of the JIA subtypes except polyarticular rheumatoid factor (RF)-positive JIA, and were particularly more likely to develop the extended oligoarticular and psoriatic subtypes. A higher frequency of enthesitis-related JIA was observed among patients of Asian origin, while those of black origin or native North American origin were more likely to develop polyarticular RF-positive JIA. CONCLUSION In this multiethnic cohort, European descent was associated with a significantly increased risk of developing JIA, and the distribution of JIA subtypes differed significantly across ethnic groups.
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Affiliation(s)
- R K Saurenmann
- Zurich University Children's Hospital, Zurich, Switzerland.
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Len CA, Terreri MT, Puccini RF, Wechsler R, Silva EK, Oliveira LM, Barbosa CMP, Pedroso GC, Hilário MOE. Development of a tool for early referral of children and adolescents with signs and symptoms suggestive of chronic arthropathy to pediatric rheumatology centers. ACTA ACUST UNITED AC 2006; 55:373-7. [PMID: 16739205 DOI: 10.1002/art.21983] [Citation(s) in RCA: 16] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/26/2022]
Abstract
OBJECTIVE To develop an easy, time-efficient tool to identify children and adolescents with signs and symptoms suggestive of chronic arthropathies, and to evaluate its interobserver reproducibility and reliability. METHODS The instrument used standardized techniques as required for the development of health-related instruments, targeting parents of apparently healthy children and adolescents ages 1-16 years. A multidisciplinary team was involved in the design of the instrument. RESULTS Each health professional generated 10-15 questions addressing musculoskeletal complaints that they considered to be the most relevant. A total of 60 questions were listed. During the reduction step, each health professional scored questions from 1 to 4 according to the question's relevance. The tool comprised 12 questions and was administered to the parents of 3 groups: patients with juvenile idiopathic arthritis (JIA; n = 48), children with diffuse musculoskeletal pain (n = 39), and a healthy control group (n = 42). The JIA group achieved the highest scores, followed by the diffuse musculoskeletal pain group and the control group. Nine (18.7%) of 48 patients with JIA and 2 (5.1%) of 39 children with musculoskeletal pain had a score of 5. The interobserver reproducibility was confirmed. All 12 questions were included in the final version of the instrument. We determined that children and adolescents with a score > or =5 should be referred for a rheumatologic evaluation (cluster analysis and logistic regression). CONCLUSION Our questionnaire seems to be a useful tool for the early detection of musculoskeletal problems in children that may need a referral for a rheumatologic evaluation.
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Affiliation(s)
- Claudio Arnaldo Len
- Escola Paulista de Medicina, Universidade Federal de São Paulo, Avenida Brigadeiro Faria Lima 1826 cj 205, São Paulo, SP CEP 01451-001, Brazil.
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