Kim Sooyoung, Park Tae Heum, Lee Seung Min, Kim Yon Hee, Cho Moon Kyun, Whang Kyu Uang, Kim Hyun-Sook
Department of Dermatology, Soonchunhyang University Seoul Hospital, Soonchunhyang University College of Medicine, Seoul, South Korea.
Department of Pathology, Soonchunhyang University Seoul Hospital, Soonchunhyang University College of Medicine, Seoul, South Korea.
Dermatol Ther. 2020 May;33(3):e13378. doi: 10.1111/dth.13378. Epub 2020 Apr 15.
Scleromyxedema is a rare connective tissue disorder characterized by a generalized lichenoid eruption and sclerodermoid induration with histologic features of dermal mucin deposition. A 44-year-old man presented with a 3-year history of generalized progressive skin thickening and sclerosis. He had diffuse skin-colored to erythematous firm papules coalescing into indurated plaques over his whole body. He had been diagnosed with scleromyxedema from a skin biopsy with monoclonal gammopathy of undetermined significance (MGUS) at another tertiary hospital 3 years earlier. He had been treated with systemic corticosteroids and methotrexate, but his systemic symptoms (dyspnea, dysphagia, skin swelling, and induration) had worsened over the past year, so he visited our clinic seeking further evaluation and management. The patient received high-dose intravenous immunoglobulin (IVIG) therapy once a month in combination with systemic corticosteroids. After three courses of IVIG, his cutaneous symptoms and dyspnea had improved dramatically. Herein we report a case of scleromyxedema with systemic involvement with significant improvement following IVIG therapy.
硬化性黏液水肿是一种罕见的结缔组织疾病,其特征为全身性苔藓样疹和硬皮病样硬结,组织学表现为真皮黏蛋白沉积。一名44岁男性,有3年全身性进行性皮肤增厚和硬化病史。他全身有弥漫性肤色至红斑性坚实丘疹,融合成硬结性斑块。3年前他在另一家三级医院经皮肤活检诊断为硬化性黏液水肿,伴有意义未明的单克隆丙种球蛋白病(MGUS)。他曾接受全身糖皮质激素和甲氨蝶呤治疗,但在过去一年中,他的全身症状(呼吸困难、吞咽困难、皮肤肿胀和硬结)有所加重,因此前来我院寻求进一步评估和治疗。该患者每月接受一次大剂量静脉注射免疫球蛋白(IVIG)治疗,并联合全身糖皮质激素。经过三个疗程的IVIG治疗后,他的皮肤症状和呼吸困难有了显著改善。在此我们报告一例伴有全身受累的硬化性黏液水肿病例,IVIG治疗后有明显改善。