出生时首次出现局限性硬皮病:硬皮病作为先天性皮肤病变的鉴别诊断

IF 0.3 Q4 PEDIATRICS Journal of Child Science Pub Date : 2022-01-01 DOI:10.1055/s-0041-1741055
E. Memar, Moeinadin Safavi, M. Moradinejad, V. Ziaee
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引用次数: 1

摘要

局限性硬皮病是一种罕见的自身免疫性疾病,其特征是皮肤和下层组织纤维化,不累及血管或内脏。它通常影响儿童在童年后期,早期表现为局限性硬皮病在婴儿期是罕见的。在目前的研究中,我们报告了一名出生时出现局限性硬皮病相关表现的儿童。一个2天大的男婴在他的左大腿上出现了一个坚硬、红斑和轻微的色素斑块,导致患足直径改变和左膝挛缩。在7个月大的时候,他被转到我们的风湿病诊所,生长发育正常。实验室研究,包括尿液和血液高效液相色谱分析,抗核抗体,抗拓扑异构酶I和风湿病因子,在正常范围内。在眼科会诊期间没有发现眼部受累的迹象。皮肤活检显示轻度棘层增生和胶原束,取代了汗腺周围的脂肪。最后诊断为局限性硬皮病。治疗开始于口服强的松龙,口服甲氨蝶呤(MTX)和秋水仙碱。治疗3个月后,皮肤病变停止进展。类固醇在6个月内逐渐减少,而MTX和秋水仙碱继续使用2年。婴儿早期的局限性硬皮病是一种罕见的疾病,但对于出生时身体上有红斑和坚硬病变的婴儿,应将其视为一种鉴别,因为早期治疗可以预防未来的并发症。
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The First Presentation of Localized Scleroderma at Birth: Scleroderma as a Differential Diagnosis of Congenital Skin Lesion
Localized scleroderma is an uncommon autoimmune disease characterized by fibrosis of the skin and underlying tissue without involvement of blood vessels or internal organs. It usually affects children during later childhood, and early presentation of localized scleroderma during infancy is rare. In the current study, we report a child with localized scleroderma-related presentations occurring at birth. A 2-day-old male neonate presented with a firm, erythematous, and slightly pigmented plaque on his left thigh, leading to a change in the diameter of the affected foot and contracture of the left knee. At the age of 7 months, he was referred to our rheumatology clinic with normal growth and development. Laboratory studies, including urine and blood high-performance liquid chromatography assay, antinuclear antibodies, antitopoisomerase I, and rheumatic factor, were in the normal range. No signs of ocular involvement were noted during ophthalmological consultation. Skin biopsy showed mild acanthosis and collagen bundles, which replaced the fat around the sweat glands. A final diagnosis of localized scleroderma was made. Treatment was started with oral prednisolone, oral methotrexate (MTX), and colchicine. The skin lesion stopped progressing after 3 months of treatment. Steroid was then tapered over 6 months, while MTX and colchicine were continued for 2 years. Localized scleroderma during early infancy is a rare disease, but it should be considered as a differential in infants with erythematous and firm lesions on their body at birth because early treatment can prevent future complications.
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