Tan Chou Luan, Yahaya Muhammad Hafizuddin, Ahmad Noor Shahrazat, Lim Chong Hong
Rheumatology Unit, Department of Internal Medicine, Hospital Sultanah Bahiyah, Alor Setar, Malaysia.
Department of Internal Medicine, Hospital Sultanah Bahiyah, Alor Setar, Malaysia.
BMJ Case Rep. 2020 Mar 12;13(3):e233330. doi: 10.1136/bcr-2019-233330.
An 18-year-old male adolescent presented with prolonged high spiking temperature, photosensitive rash, oral ulcers and reduced hearing bilaterally of recent onset. Examination revealed malar rash, vasculitis rash over bilateral palms and soles, oral and buccal ulcers, palpable posterior auricular and inguinal lymph nodes, and reduced hearing bilaterally. His further investigations noted pancytopaenia, elevated transaminases, hyperferritinaemia, low C3 and C4 levels, positive antinuclear antibody, double-stranded DNA and direct Coombs test, while bone marrow aspiration revealed active phagocytic activity suggestive of hemophagocytic syndrome. We made a diagnosis of systemic lupus erythematosus with macrophage activation syndrome. We treated him with pulse intravenous methylprednisolone and his condition improved drastically. Temperature resolved on the next day after intravenous methylprednisolone; bilateral sensorineural hearing loss improved to near-normal hearing after treatment. He remained well during follow-up with a tapering dose of prednisolone.
一名18岁男性青少年近期出现持续的高热、光敏性皮疹、口腔溃疡以及双侧听力下降。检查发现有蝶形皮疹、双侧手掌和足底的血管炎皮疹、口腔和颊部溃疡、可触及的耳后和腹股沟淋巴结,以及双侧听力下降。进一步检查发现全血细胞减少、转氨酶升高、铁蛋白血症、C3和C4水平降低、抗核抗体、双链DNA及直接抗人球蛋白试验阳性,而骨髓穿刺显示有活跃的吞噬活性,提示噬血细胞综合征。我们诊断为系统性红斑狼疮合并巨噬细胞活化综合征。我们用静脉注射甲泼尼龙冲击治疗,他的病情显著改善。静脉注射甲泼尼龙后的第二天体温恢复正常;治疗后双侧感音神经性听力损失改善至接近正常听力。在逐渐减量泼尼松龙的随访期间,他情况良好。