César Thibaut, Giovannini Diane, Deroux Alban, Bouillet Laurence, Nugues Frédérique, Carbasse Aurélia, Koné-Paut Isabelle, Borocco Charlotte, Pietrement Christine, Bader-Meunier Brigitte, Quartier Pierre, Laurent Audrey, Giani Teresa, Brück Normi, Calzada-Hernàndez Joan, Pagnier Anne, Kevorkian-Verguet Charlotte
Department of Pediatrics, Grenoble-Alpes University Hospital, Grenoble, France.
Pathology Department, Grenoble-Alpes University Hospital, Grenoble, France.
Eur J Pediatr. 2025 Jan 28;184(2):156. doi: 10.1007/s00431-025-05981-5.
The purpose of this study was to identify pediatric eosinophilic fasciitis, which is an extremely rare condition, in order to describe their clinical, paraclinical, and therapeutic characteristics. We made a call for observations via societies for pediatric rheumatology in France and surrounding countries and collected clinical and paraclinical data of the cases fulfilling the diagnostic criteria. Eight patients under 18 years of age with confirmed eosinophilic fasciitis followed between April 2004 and July 2022 in France, Germany, Italy, and Spain were included. The median age of onset of symptoms was 8.7 years (range 3 to 12.6). All patients had skin and joint involvement at diagnosis. Eosinophilia was present at diagnosis in 5/8 patients and 5/7 patients presented hypergammaglobulinemia. All the patients had an MRI, and in 6, we observed thickened fascia with a T2 hypersignal. Five patients had undergone a full-thickness biopsy showing a polymorphic lymphoplasmacytic infiltrate of the fascia in all and the presence of eosinophils in 4 of them. All the patients were treated with corticosteroids with variable regimens and all received at least an immunosuppressant. Conclusion: To our knowledge, this is the largest pediatric series of eosinophilic fasciitis. The clinical and paraclinical presentation seems similar to that of adults except for a form that appears to be distinguished with isolated joint contractures, and hypergammaglobulinemia which appears to be more frequent in children. Dermatological and pathological expertise and MRI are key elements of the diagnosis. The most consensual treatment includes physiotherapy, prolonged corticosteroid therapy, and methotrexate as first-line therapy. What is Known • Eosinophilic fasciitis is a rare condition, especially in children with approximately 60 reported pediatric cases. • Some pediatric specificities tend to emerge from some reports. What is New • Possible isolated joints contractures and more frequent hypergammaglobulinemia seem to characterize pediatric eosinophilic fasciitis in comparison with adult forms. • The most frequently used first-line therapy combines physiotherapy, corticosteroids and methrotrexate.
本研究的目的是识别儿童嗜酸性筋膜炎(一种极为罕见的疾病),以描述其临床、副临床和治疗特征。我们通过法国及周边国家的儿童风湿病学会征集病例,并收集符合诊断标准的病例的临床和副临床数据。纳入了2004年4月至2022年7月期间在法国、德国、意大利和西班牙确诊为嗜酸性筋膜炎的8名18岁以下患者。症状出现的中位年龄为8.7岁(范围3至12.6岁)。所有患者在诊断时均有皮肤和关节受累。8例患者中有5例在诊断时有嗜酸性粒细胞增多,7例患者中有5例出现高球蛋白血症。所有患者均接受了MRI检查,其中6例观察到筋膜增厚伴T2高信号。5例患者接受了全层活检,所有患者均显示筋膜有多形性淋巴浆细胞浸润,其中4例有嗜酸性粒细胞。所有患者均接受了不同方案的皮质类固醇治疗,且均至少接受了一种免疫抑制剂治疗。结论:据我们所知,这是最大的儿童嗜酸性筋膜炎系列研究。除了一种以孤立关节挛缩为特征的类型以及儿童中似乎更常见的高球蛋白血症外,其临床和副临床表现似乎与成人相似。皮肤科和病理学专业知识以及MRI是诊断的关键要素。最一致认可的治疗方法包括物理治疗、长期皮质类固醇治疗以及甲氨蝶呤作为一线治疗。已知信息:嗜酸性筋膜炎是一种罕见疾病,尤其是在儿童中,约有60例儿童病例报告。一些报告中出现了一些儿童特有的情况。新发现:与成人形式相比,可能的孤立关节挛缩和更频繁的高球蛋白血症似乎是儿童嗜酸性筋膜炎的特征。最常用的一线治疗方法是物理治疗、皮质类固醇和甲氨蝶呤联合使用。