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面部毛细血管扩张——新生儿红斑狼疮的一种罕见并发症:一例报告

Facial telangiectasia-an unusual complication of neonatal lupus erythematosus: report of one case.

作者信息

Lin Sheng-Chieh, Shyur Shyh-Dar, Wu Jiunn-Yi, Huang Li-Hsin

机构信息

Department of Pediatrics, Mackay Memorial Hospital, Taipei, Taiwan.

出版信息

Acta Paediatr Taiwan. 2004 Jul-Aug;45(4):246-8.

PMID:15624375
Abstract

Neonatal lupus erythematosus (NLE) is an uncommon passive autoimmune disease caused by transplacental passage of anti-Ro/SSA and/or anti-La/SSB or anti-U1RNP maternal autoantibodies. Common clinical manifestations include cutaneous lupus lesions, cardiac disease, notably congenital heart block, hematologic abnormalities, and hepatobiliary disease. The cutaneous lesions of NLE are usually transient, disappearing about six months after birth when maternal antibodies disappear from the infant's circulation. Persistent telangiectasia is a rare complication of NLE. We report a 3-year-old female who had cutaneous lupus with persistent facial telangiectasias over the frontal area. She was diagnosed with NLE at 2 months of age. Her findings then included cutaneous lupus, hemolytic anemia, a high titer of antinuclear antibodies (1: 640) with a speckled pattern, positive anti-Ro/SSA and anti-La/SSB antibodies, and absence of anti-dsDNA antibodies. Her mother had systemic lupus erythematosus with the presence of high titer of antinuclear antibodies (1: 1260) with a speckled pattern and positive anti-Ro/SSA and anti-La/SSB antibodies. The child's cutaneous lupus and hemolytic anemia disappeared at 6 months of age, but the telangiectasia persisted.

摘要

新生儿红斑狼疮(NLE)是一种罕见的被动自身免疫性疾病,由抗Ro/SSA和/或抗La/SSB或抗U1RNP母体自身抗体经胎盘传递引起。常见临床表现包括皮肤狼疮病变、心脏病,尤其是先天性心脏传导阻滞、血液学异常和肝胆疾病。NLE的皮肤病变通常是短暂的,在出生后约六个月母体抗体从婴儿循环中消失时消失。持续性毛细血管扩张是NLE的一种罕见并发症。我们报告一名3岁女性,其额部有持续性面部毛细血管扩张的皮肤狼疮。她在2个月大时被诊断为NLE。当时她的检查结果包括皮肤狼疮、溶血性贫血、高滴度(1:640)斑点型抗核抗体、抗Ro/SSA和抗La/SSB抗体阳性,以及抗双链DNA抗体阴性。她的母亲患有系统性红斑狼疮,有高滴度(1:1260)斑点型抗核抗体以及抗Ro/SSA和抗La/SSB抗体阳性。患儿的皮肤狼疮和溶血性贫血在6个月大时消失,但毛细血管扩张持续存在。

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