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新生儿狼疮患儿及其未患病同胞的长期随访

Long-term followup of children with neonatal lupus and their unaffected siblings.

作者信息

Martin Victor, Lee Lela A, Askanase Anca D, Katholi Margaret, Buyon Jill P

机构信息

Hospital for Joint Diseases, New York University School of Medicine, New York, New York 10003, USA.

出版信息

Arthritis Rheum. 2002 Sep;46(9):2377-83. doi: 10.1002/art.10638.

Abstract

OBJECTIVE

To determine in a longitudinal cohort study whether children with varied manifestations of neonatal lupus or their unaffected siblings later develop autoantibodies and/or rheumatic diseases.

METHODS

To obtain information on the health of children ages >or=8 years who had manifestations of neonatal lupus (affected group) and their unaffected siblings (unaffected group), questionnaires were sent to mothers (with anti-SSA/Ro and/or anti-SSB/La antibodies) who were enrolled in the National Institute of Arthritis and Musculoskeletal and Skin Diseases/Hospital for Joint Diseases Research Registry for Neonatal Lupus. Children of healthy mothers referred by the Registry enrollees comprised the control group. Further data were provided by review of medical records.

RESULTS

Fifty-five mothers enrolled in the Registry returned questionnaires on 49 children with neonatal lupus and their 45 unaffected siblings. Six children with definite rheumatic/autoimmune diseases were identified: 2 with juvenile rheumatoid arthritis, 1 with Hashimoto thyroiditis, 1 with psoriasis and iritis, 1 with diabetes mellitus and psoriasis, and 1 with congenital hypothyroidism and nephrotic syndrome. All had neonatal lupus, and their mothers had manifestations of autoimmune diseases (Sjögren's syndrome in 4, systemic lupus erythematosus/Sjögren's syndrome in 1, and undifferentiated autoimmune disease in 1). Antinuclear antibodies were present in 4 of 55 sera tested (2 of 33 affected children and 2 of 22 unaffected children). No serum contained antibodies reactive with SSA/Ro or SSB/La antigens.

CONCLUSION

These data suggest that children with neonatal lupus require continued followup, especially prior to adolescence and if the mother herself has an autoimmune disease. While there was no apparent increased risk of systemic lupus erythematosus, the development of some form of autoimmune disease (systemic or organ-specific) in early childhood may be of concern. During adolescence and young adulthood, individuals with neonatal lupus and their unaffected siblings do not appear to have an increased risk of developing systemic rheumatic diseases.

摘要

目的

在一项纵向队列研究中,确定患有各种新生儿狼疮表现的儿童或其未受影响的兄弟姐妹日后是否会出现自身抗体和/或风湿性疾病。

方法

为获取有关年龄≥8岁、有新生儿狼疮表现的儿童(患病组)及其未受影响的兄弟姐妹(未患病组)的健康信息,向参加国立关节炎、肌肉骨骼和皮肤病研究所/关节疾病医院新生儿狼疮研究登记处的母亲(有抗SSA/Ro和/或抗SSB/La抗体)发送了问卷。登记参与者推荐的健康母亲的孩子组成对照组。通过查阅病历提供了进一步的数据。

结果

登记处的55位母亲返回了有关49名患有新生儿狼疮的儿童及其45名未受影响的兄弟姐妹的问卷。确定了6名患有明确风湿性/自身免疫性疾病的儿童:2名患有幼年类风湿关节炎,1名患有桥本甲状腺炎,1名患有银屑病和虹膜炎,1名患有糖尿病和银屑病,1名患有先天性甲状腺功能减退症和肾病综合征。所有这些儿童都患有新生儿狼疮,且他们的母亲有自身免疫性疾病表现(4例为干燥综合征,1例为系统性红斑狼疮/干燥综合征,1例为未分化自身免疫性疾病)。在检测的55份血清中,4份存在抗核抗体(33名患病儿童中的2份和22名未患病儿童中的2份)。没有血清含有与SSA/Ro或SSB/La抗原反应的抗体。

结论

这些数据表明,患有新生儿狼疮的儿童需要持续随访,尤其是在青春期之前以及母亲本身患有自身免疫性疾病的情况下。虽然系统性红斑狼疮的风险没有明显增加,但儿童早期出现某种形式的自身免疫性疾病(全身性或器官特异性)可能令人担忧。在青春期和成年早期,患有新生儿狼疮的个体及其未受影响的兄弟姐妹患系统性风湿性疾病的风险似乎没有增加。

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