Hatano Taku, Takanashi Masashi, Tsuchihashi Hitoshi, Ueno Shin-Ichi, Hayashida Arisa, Tsukune Yutaka, Kanai Kazuaki, Shimo Yasushi, Hattori Nobutaka
Department of Neurology, Juntendo University Graduate School of Medicine, 2-1-1 Hongo, Bunkyo-ku, Tokyo, 113-8421, Japan.
Department of Neurology, Juntendo Koshigaya Hospital, 560 Fukuroyama, Koshigayashi, Saitama, 343-0032, Japan.
BMC Neurol. 2018 Aug 14;18(1):112. doi: 10.1186/s12883-018-1123-3.
Cutaneous and systemic plasmacytosis are skin disorders characterized by cutaneous polyclonal plasma cell infiltration accompanied by polyclonal hypergammaglobulinemia. Cutaneous plasmacytosis involvement is limited to the skin, mainly on the face and trunk, while systemic plasmacytosis also involves 2 or more organ systems. However, there have been no reports of inflammatory myositis due to plasmacytosis. Here, we report a patient with plasmacytosis who developed myalgia and easy fatigability due to inflammatory myositis.
A 54-year-old man with cutaneous plasmacytosis on the face, chest, and back complained of a history of atypical facial and lower leg pain and easy fatigability since the age of 45 years. Muscle-strength tests revealed bilateral trivial gastrocnemius weakness with myalgia. The results of routine blood analysis, including creatine kinase and thyroid function, were normal, but levels of several inflammation markers and autoantibodies were elevated. Additionally, lower leg magnetic resonance imaging and gastrocnemius muscle biopsy revealed inflammatory myositis mimicking polymyositis. His plasmacytosis, myalgia, and lower leg weakness were ameliorated by prednisolone.
The patient was diagnosed with inflammatory myositis due to plasmacytosis. Given that plasmacytosis has previously been reported to disrupt the immune status, myositis in this patient might have been associated with abnormal autoimmune inflammation. Neurologists and physicians should thus be aware that plasmacytosis might be associated with inflammatory myositis accompanied by myalgia.
皮肤和系统性浆细胞增多症是一种皮肤疾病,其特征为皮肤多克隆浆细胞浸润并伴有多克隆高球蛋白血症。皮肤浆细胞增多症仅累及皮肤,主要在面部和躯干,而系统性浆细胞增多症还累及两个或更多器官系统。然而,此前尚无浆细胞增多症导致炎性肌炎的报道。在此,我们报告一例浆细胞增多症患者,其因炎性肌炎出现肌痛和易疲劳症状。
一名54岁男性,面部、胸部和背部患有皮肤浆细胞增多症,自45岁起即有非典型面部和小腿疼痛及易疲劳病史。肌力测试显示双侧腓肠肌轻度无力并伴有肌痛。包括肌酸激酶和甲状腺功能在内的常规血液分析结果正常,但多项炎症标志物和自身抗体水平升高。此外,小腿磁共振成像和腓肠肌活检显示为类似多发性肌炎的炎性肌炎。泼尼松龙使他的浆细胞增多症、肌痛和小腿无力症状得到改善。
该患者被诊断为浆细胞增多症所致炎性肌炎。鉴于此前有报道称浆细胞增多症会破坏免疫状态,该患者的肌炎可能与异常的自身免疫炎症有关。因此,神经科医生和内科医生应意识到浆细胞增多症可能与伴有肌痛的炎性肌炎有关。