Department of General Internal Medicine, Fukushima Medical University, Fukushima, Japan.
Department of General Internal Medicine, Fukushima Medical University, Fukushima, Japan
BMJ Case Rep. 2020 Nov 4;13(11):e236283. doi: 10.1136/bcr-2020-236283.
A woman aged 45 years with a 1.5-year history of violaceous plaques on the forehead and chest presented with fever, weight loss and aggravation of the plaques. Inflammatory markers and interleukin-6 level were elevated, and superficial lymphadenopathies and splenomegaly were identified by CT scan. Immunohistochemical findings of the lymph node and the skin showed polyclonal plasmacytosis and follicular hyperplasia, leading to the diagnosis of idiopathic multicentric Castleman disease (iMCD) after human herpesvirus-8 infection was excluded. The patient was successfully treated with anti-interleukin-6 receptor antibody, tocilizumab, following relapse after prednisolone therapy.Our literature review found 11 case reports of pathologically confirmed iMCD preceded by cutaneous plasmacytosis. The median duration of asymptomatic phase with only skin lesions was 7.5 years, whereas the phase lasted only for 1.5 years in our case. iMCD can develop shortly after asymptomatic cutaneous plasmacytosis. Tocilizumab can be a treatment of choice for this type of iMCD.
一位 45 岁女性,前额和胸部有 1.5 年紫红色斑块病史,出现发热、体重减轻和斑块加重。炎症标志物和白细胞介素-6 水平升高,CT 扫描发现浅表淋巴结病和脾肿大。淋巴结和皮肤的免疫组织化学检查显示多克隆浆细胞增生和滤泡增生,在排除人类疱疹病毒-8 感染后,诊断为特发性多中心 Castleman 病(iMCD)。在泼尼松龙治疗后复发后,患者成功接受抗白细胞介素-6 受体抗体托珠单抗治疗。我们的文献复习发现 11 例经病理证实的 iMCD 病例,此前有皮肤浆细胞瘤。无症状期仅有皮肤病变的中位持续时间为 7.5 年,而在我们的病例中仅持续 1.5 年。iMCD 可在无症状性皮肤浆细胞瘤后不久发生。托珠单抗可能是这种类型 iMCD 的首选治疗方法。