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      Grisel’s syndrome in Kawasaki disease

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          Abstract

          Background

          Approximately 50–70% of patients with Kawasaki disease (KD) could present with cervical lymphadenopathy associated with deep neck inflammation, which may result in Grisel’s syndrome (GS). Given the possibility of neurological impairment owing to GS, it is important to understand the disease profile in KD. Therefore, we carried out this study to investigate this possible complication of KD, with the aim of improving pediatricians’ recognition and awareness.

          Methods

          Patients with KD complicated by GS in our hospital were retrospectively recruited for our study. The profiles of patients with GS ( n = 10) were compared to those patients without GS ( n = 1254). All the available literature describing these complications of KD was reviewed.

          Results

          The incidence of GS in KD was 0.6% in our population. Compared to patients without GS, KD patients with GS were older, presented with a significantly lower male:female ratio, and a higher incidence of cervical lymphadenopathy, a higher level of neutrophil count, and erythrocyte sedimentation rate. Ten articles reporting 14 KD patients with GS were reviewed. Of the total 24 patients, GS affected 7 males and 17 females, aged from 3.5 to 9 years old. Encouragingly, no delayed diagnosis and treatment of KD was found, and all patients received conservative therapy for GS, without intravenous immunoglobulin resistance, coronary artery lesions, and neurological impairment.

          Conclusions

          GS is a rare complication of KD with an incidence of 0.6%, predominantly affecting older, female children. The overall outcome of this disorder in KD was satisfactory with conservative therapy. Pediatricians, especially pediatric surgeons, should recognize and be aware of this possible complication of KD to avoid misdiagnosis and overtreatment.

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          Most cited references32

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          Epidemiologic Features of Kawasaki Disease in Japan: Results of the 2009–2010 Nationwide Survey

          Background Although the number of patients and incidence rate of Kawasaki disease (KD) are increasing in Japan, the most recent epidemiologic features of KD are not known. Methods The 21st nationwide survey of KD was conducted in 2011 and included patients treated for the disease in 2009 and 2010. Hospitals specializing in pediatrics, and hospitals with a total of 100 or more beds and a pediatric department, were asked to report all patients with KD during the 2 survey years. Results A total of 1445 departments and hospitals reported 23 730 KD patients (10 975 in 2009 and 12 755 in 2010): 13 515 boys and 10 215 girls. The annual incidence rates were 206.2 and 239.6 per 100 000 children aged 0 to 4 years in 2009 and 2010, respectively; the 2010 rate was the highest ever reported in Japan. Monthly number of patients peaked during winter to spring months; lower peaks were noted during summer months. However, the seasonal patterns in 2009 and 2010 differed from those of previous years. The age-specific incidence rate had a monomodal distribution, with a peak during the latter half of the year of birth. The prevalences of cardiac lesions during acute KD and cardiac sequelae were higher among infants and older age groups. Despite a decrease in prevalence, the proportion of patients with giant coronary aneurysms—the most severe sequela of KD—did not substantially decrease. Conclusions The incidence rate and number of patients with KD continue to increase in Japan.
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            Epidemiologic Features of Kawasaki Disease in Shanghai From 2008 Through 2012.

            This study was to investigate the epidemiologic trends of Kawasaki disease (KD) and coronary arterial lesions (CALs) in Shanghai from 2008 through 2012.
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              Lymph-node-first presentation of Kawasaki disease compared with bacterial cervical adenitis and typical Kawasaki disease.

              To identify characteristics differentiating the node-first presentation of Kawasaki disease (NFKD) from bacterial cervical lymphadenitis (BCL) and typical Kawasaki disease (KD).
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                Author and article information

                Contributors
                805101396@qq.com
                Journal
                Orphanet J Rare Dis
                Orphanet J Rare Dis
                Orphanet Journal of Rare Diseases
                BioMed Central (London )
                1750-1172
                11 September 2020
                11 September 2020
                2020
                : 15
                : 246
                Affiliations
                [1 ]GRID grid.461863.e, ISNI 0000 0004 1757 9397, Department of Pediatric Cardiology, , West China Second University Hospital, Sichuan University, ; Chengdu, 610041 Sichuan China
                [2 ]GRID grid.13291.38, ISNI 0000 0001 0807 1581, Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education Chengdu, ; Chengdu, 610041 Sichuan China
                [3 ]GRID grid.461863.e, ISNI 0000 0004 1757 9397, Key Laboratory of Development and Diseases of Women and Children of Sichuan Province, , West China Second University Hospital, Sichuan University, ; Chengdu, 610041 Sichuan China
                [4 ]GRID grid.13291.38, ISNI 0000 0001 0807 1581, The Cardiac development and early intervention unit, , West China Institute of Women and Children’s Health, West China Second University Hospital, Sichuan University, ; Chengdu, 610041 Sichuan China
                [5 ]GRID grid.13291.38, ISNI 0000 0001 0807 1581, West China Medical School of Sichuan University, ; Chengdu, 610041 Sichuan China
                Author information
                http://orcid.org/0000-0001-5165-8273
                Article
                1535
                10.1186/s13023-020-01535-0
                7488729
                32917253
                3f773f9d-0341-4532-a6bb-913d650b7e0d
                © The Author(s) 2020

                Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

                History
                : 16 February 2020
                : 7 September 2020
                Funding
                Funded by: FundRef http://dx.doi.org/10.13039/501100001809, National Natural Science Foundation of China;
                Award ID: No. 81741026 and No. 81571515
                Award ID: 81800288
                Award Recipient :
                Funded by: Science‐technology Support Plan Projects in Sichuan province
                Award ID: 2019YFS0243
                Award ID: 2017SZ0117
                Award Recipient :
                Funded by: Technology Innovation Research and Development Project of Chengdu Science and Technology Bureau
                Award ID: No 2018-YF05-00249-SN
                Award Recipient :
                Categories
                Research
                Custom metadata
                © The Author(s) 2020

                Infectious disease & Microbiology
                atlantoaxial subluxation,cervical lymphadenopathy,neurological impairment,coronary artery lesions,children

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