Coexisting Sacroiliac Arthritis and Chronic Nonbacterial Osteomyelitis in an Adolescent with Ehlers-Danlos Syndrome: A Case Report and Treatment Success.

IF 1 Q3 MEDICINE, GENERAL & INTERNAL
Joanna Ożga, Elżbieta Mężyk, Wojciech Kmiecik, Wadim Wojciechowski, Zbigniew Żuber
{"title":"Coexisting Sacroiliac Arthritis and Chronic Nonbacterial Osteomyelitis in an Adolescent with Ehlers-Danlos Syndrome: A Case Report and Treatment Success.","authors":"Joanna Ożga, Elżbieta Mężyk, Wojciech Kmiecik, Wadim Wojciechowski, Zbigniew Żuber","doi":"10.12659/AJCR.943579","DOIUrl":null,"url":null,"abstract":"<p><p>BACKGROUND Chronic nonbacterial osteomyelitis (CNO) is a multifocal autoinflammatory bone disease mainly affecting children and adolescents. Sacroiliitis is an inflammation of the sacroiliac joint, diagnosed with the use of musculoskeletal MRI due to its ability to visualize active inflammatory lesions. Ehlers-Danlos syndrome (EDS) is non-inflammatory hereditary disorder of connective tissue. Here, we report the case of a 17.5-year-old female patient with classical EDS and long-term course of the CNO with coexistence of sacroiliac arthritis. CASE REPORT On admission, a patient with CNO reported pain in the scapula, thoracic spine, shoulders, and iliac region, with morning stiffness present for 5 months. Physical examination revealed knee and elbow joint hyperextension, hypermobility of the phalanges, increased range of motion of the hip joints, and the presence of reticular rash on the face. In the laboratory blood tests, minor leukocytosis was reported. During hospitalization, a whole-body MRI was performed, detecting bone marrow edema in the Th3, Th4, and Th7 vertebral bodies and the head of seventh rib on the left side, as well as bilaterally in the sacroiliac joints. The patient was diagnosed with sacroiliitis and EDS and successfully treated with risedronate sodium, methotrexate with folic acid, sulfasalazine, and meloxicam, achieving CNO remission and reduced severity of axial skeleton pain. CONCLUSIONS The coexistence of these 3 diseases - CNO, sacroiliac arthritis, and EDS - in the same patient is rare and requires interphysician collaboration to determine the correct diagnosis and subsequently arrange multi-speciality therapeutic management to achieve remission.</p>","PeriodicalId":39064,"journal":{"name":"American Journal of Case Reports","volume":null,"pages":null},"PeriodicalIF":1.0000,"publicationDate":"2024-09-22","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11426177/pdf/","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"American Journal of Case Reports","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.12659/AJCR.943579","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"MEDICINE, GENERAL & INTERNAL","Score":null,"Total":0}
引用次数: 0

Abstract

BACKGROUND Chronic nonbacterial osteomyelitis (CNO) is a multifocal autoinflammatory bone disease mainly affecting children and adolescents. Sacroiliitis is an inflammation of the sacroiliac joint, diagnosed with the use of musculoskeletal MRI due to its ability to visualize active inflammatory lesions. Ehlers-Danlos syndrome (EDS) is non-inflammatory hereditary disorder of connective tissue. Here, we report the case of a 17.5-year-old female patient with classical EDS and long-term course of the CNO with coexistence of sacroiliac arthritis. CASE REPORT On admission, a patient with CNO reported pain in the scapula, thoracic spine, shoulders, and iliac region, with morning stiffness present for 5 months. Physical examination revealed knee and elbow joint hyperextension, hypermobility of the phalanges, increased range of motion of the hip joints, and the presence of reticular rash on the face. In the laboratory blood tests, minor leukocytosis was reported. During hospitalization, a whole-body MRI was performed, detecting bone marrow edema in the Th3, Th4, and Th7 vertebral bodies and the head of seventh rib on the left side, as well as bilaterally in the sacroiliac joints. The patient was diagnosed with sacroiliitis and EDS and successfully treated with risedronate sodium, methotrexate with folic acid, sulfasalazine, and meloxicam, achieving CNO remission and reduced severity of axial skeleton pain. CONCLUSIONS The coexistence of these 3 diseases - CNO, sacroiliac arthritis, and EDS - in the same patient is rare and requires interphysician collaboration to determine the correct diagnosis and subsequently arrange multi-speciality therapeutic management to achieve remission.

一名患有埃勒斯-丹洛斯综合征的青少年同时患有骶髂关节炎和慢性非细菌性骨髓炎:病例报告与成功治疗
背景 慢性非细菌性骨髓炎(CNO)是一种多灶性自身炎症性骨病,主要影响儿童和青少年。骶髂关节炎是一种骶髂关节炎症,可通过肌肉骨骼核磁共振成像诊断,因为它能显示活动性炎症病灶。埃勒斯-丹洛斯综合征(EDS)是一种非炎症性遗传性结缔组织疾病。在此,我们报告了一例 17.5 岁女性患者的病例,她患有典型的 EDS 和长期的 CNO 病程,并同时伴有骶髂关节炎。病例报告 一名 CNO 患者入院时报告肩胛骨、胸椎、肩膀和髂骨区域疼痛,晨僵已持续 5 个月。体格检查显示膝关节和肘关节过度伸展,趾骨活动度过大,髋关节活动范围增大,面部出现网状皮疹。在实验室血液检查中,报告了轻微的白细胞增多。住院期间,患者接受了全身核磁共振成像检查,结果发现左侧Th3、Th4、Th7椎体和第七根肋骨头以及双侧骶髂关节出现骨髓水肿。患者被诊断为骶髂关节炎和 EDS,并成功接受了利塞膦酸钠、含叶酸的甲氨蝶呤、柳氮磺胺吡啶和美洛昔康的治疗,获得了 CNO 缓解并减轻了轴性骨骼疼痛的程度。结论 同一名患者同时患有 CNO、骶髂关节炎和 EDS 这三种疾病的情况非常罕见,需要医生之间通力合作,确定正确的诊断,然后安排多专科治疗,以达到缓解的目的。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 求助全文
来源期刊
American Journal of Case Reports
American Journal of Case Reports Medicine-Medicine (all)
CiteScore
1.80
自引率
0.00%
发文量
599
期刊介绍: American Journal of Case Reports is an international, peer-reviewed scientific journal that publishes single and series case reports in all medical fields. American Journal of Case Reports is issued on a continuous basis as a primary electronic journal. Print copies of a single article or a set of articles can be ordered on demand.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信