Use of tocilizumab to treat arthritis associated with mixed connective tissue disease complicated by ovarian teratoma: a case report.

IF 1.2 4区 医学 Q2 MEDICINE, GENERAL & INTERNAL Journal of Nippon Medical School Pub Date : 2024-06-18 DOI:10.1272/jnms.JNMS.2025_92-303
Haruka Ota, Toru Igarashi, Ryosuke Matsui, Hikaru Takeshita, Koji Hashimoto, Masaki Miyao, Norio Motoda, Tsubasa Takahashi, Jun Hayakawa, Makoto Migita, Yasuhiko Itoh
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Abstract

Mixed connective tissue disease (MCTD) is characterized by mixed features of systemic lupus erythematosus, systemic sclerosis, and polymyositis/dermatomyositis and is rare in children. Here, we report a case of MCTD in a 10-year-old girl who presented at our hospital with arthralgia, Raynaud's phenomenon, and fatigue. Blood tests were positive for anti-U1-ribonucleoprotein (RNP) antibodies and for rheumatoid factors (RFs) IgG-RF and anti-galactose-deficient IgG. Levels of myogenic enzymes and hypergammaglobulinemia were elevated. Macrophages were prominent in bone marrow, with scattered phagocytic macrophages. MCTD was diagnosed based on the patient's symptoms and laboratory findings. Methylprednisolone pulse therapy combined with oral tacrolimus was administered, which led to resolution of symptoms. Three months after pulse therapy, arthralgia worsened and methotrexate was administered. Arthralgia improved but did not resolve. Magnetic resonance imaging performed to investigate the hip pain revealed a mature ovarian teratoma, which was surgically removed. Because the pain persisted and interfered with her daily life, she was treated with tocilizumab for joint pain relief, which decreased the pain level. Tocilizumab is a candidate for additional treatment of juvenile idiopathic arthritis-like arthritis associated with childhood-onset MCTD.

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使用托西珠单抗治疗卵巢畸胎瘤并发混合结缔组织病相关关节炎:病例报告。
混合性结缔组织病(MCTD)的特征是系统性红斑狼疮、系统性硬化症和多发性肌炎/皮肌炎的混合症状,在儿童中非常罕见。在此,我们报告了一例 10 岁女孩的 MCTD 病例,她因关节痛、雷诺现象和乏力到我院就诊。血检结果显示抗 U1 核糖核蛋白(RNP)抗体、类风湿因子(RFs)IgG-RF 和抗半乳糖缺乏 IgG 阳性。肌酶和高丙种球蛋白血症水平升高。骨髓中巨噬细胞突出,并伴有散在的吞噬性巨噬细胞。根据患者的症状和实验室检查结果,确诊为 MCTD。患者接受了甲基强的松龙脉冲疗法,同时口服他克莫司,症状有所缓解。脉冲疗法三个月后,关节痛加重,于是使用了甲氨蝶呤。关节痛有所改善,但没有缓解。为检查髋部疼痛而进行的磁共振成像检查发现了一个成熟的卵巢畸胎瘤,并进行了手术切除。由于疼痛持续存在,影响了她的日常生活,她接受了托珠单抗治疗以缓解关节疼痛,疼痛程度有所减轻。托西珠单抗是治疗与儿童期发病的 MCTD 相关的幼年特发性关节炎样关节炎的一种候选药物。
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来源期刊
Journal of Nippon Medical School
Journal of Nippon Medical School MEDICINE, GENERAL & INTERNAL-
CiteScore
1.80
自引率
10.00%
发文量
118
期刊介绍: The international effort to understand, treat and control disease involve clinicians and researchers from many medical and biological science disciplines. The Journal of Nippon Medical School (JNMS) is the official journal of the Medical Association of Nippon Medical School and is dedicated to furthering international exchange of medical science experience and opinion. It provides an international forum for researchers in the fields of bascic and clinical medicine to introduce, discuss and exchange thier novel achievements in biomedical science and a platform for the worldwide dissemination and steering of biomedical knowledge for the benefit of human health and welfare. Properly reasoned discussions disciplined by appropriate references to existing bodies of knowledge or aimed at motivating the creation of such knowledge is the aim of the journal.
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