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The bm12 Inducible Model of Systemic Lupus Erythematosus (SLE) in C57BL/6 Mice
Published on: November 1, 2015
Long-term followup of children with neonatal lupus and their unaffected siblings
Victor Martin1, Lela A Lee, Anca D Askanase
1Hospital for Joint Diseases, New York University School of Medicine, New York, New York 10003, USA.
Insights
Children with neonatal lupus may develop autoimmune diseases, requiring ongoing monitoring. Unaffected siblings and those with neonatal lupus show no increased risk of systemic rheumatic diseases in adolescence.
Area of Science:
- Pediatrics
- Rheumatology
- Immunology
Background:
- Neonatal lupus (NL) is a passively acquired condition due to maternal autoantibodies.
- Long-term outcomes for children with NL and their siblings are not well-established.
Purpose of the Study:
- To investigate the long-term development of autoantibodies and rheumatic diseases in children with NL and their unaffected siblings.
Main Methods:
- A longitudinal cohort study involving mothers enrolled in a Neonatal Lupus Registry.
- Questionnaires were sent to mothers of children with NL and their unaffected siblings (aged >or=8 years).
- Medical records were reviewed for further data.
Main Results:
- Six children (all with NL) developed definite rheumatic/autoimmune diseases, including juvenile rheumatoid arthritis, Hashimoto thyroiditis, psoriasis with iritis, diabetes mellitus with psoriasis, and congenital hypothyroidism with nephrotic syndrome.
- Mothers of affected children had autoimmune diseases (Sjögren's syndrome, SLE/Sjögren's, or undifferentiated autoimmune disease).
- Antinuclear antibodies were detected in 4 of 55 children; SSA/Ro or SSB/La antibodies were not found.
Conclusions:
- Children with NL warrant continued follow-up, particularly before adolescence and if the mother has an autoimmune disease.
- While not increasing the risk of systemic lupus erythematosus, NL may be associated with early-onset autoimmune diseases.
- No increased risk of systemic rheumatic diseases was observed in adolescence and young adulthood for individuals with NL or their siblings.
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.