Disease progression in mothers of children enrolled in the Research Registry for Neonatal Lupus
- 1 June 2009
- journal article
- research article
- Published by Elsevier in Annals of the Rheumatic Diseases
- Vol. 68 (6) , 828-835
- https://doi.org/10.1136/ard.2008.088054
Abstract
Objective: To evaluate autoimmune disease progression in asymptomatic and pauci-symptomatic mothers of children with neonatal lupus (NL). Methods: Clinical information on mothers enrolled in the Research Registry for NL (RRNL) was obtained from medical records. Genotyping was performed for −308A/G tumour necrosis factor (TNF)α, 869T/C transforming growth factor (TGF)β and −889C/T interleukin (IL)1α. Results: Of the 321 mothers enrolled, 229 had at least 6 months of follow-up. Of the 51 mothers who were asymptomatic at the NL child’s birth, 26 progressed: 12 developed pauci-undifferentiated autoimmune syndrome (pauci-UAS), 2 poly-UAS, 7 SS, 4 SLE and 1 SLE/SS. The median time to develop any symptom was 3.15 years. Of the 37 mothers classified as pauci-UAS at the NL child’s birth, 16 progressed: 5 developed poly-UAS, 6 Sjögren syndrome (SS), 4 systemic lupus erythematosus (SLE) and 1 SLE/SS. Of the pauci-UAS mothers enrolled within 1 year, the median time to progression was 6.7 years. Four mothers developed lupus nephritis (two asymptomatic, two pauci-UAS). The probability of an asymptomatic mother developing SLE by 10 years was 18.6%, and developing probable/definite SS was 27.9%. NL manifestations did not predict disease progression in an asymptomatic mother. Mothers with anti-Sjögren syndrome A antigen (SSA/)Ro and anti-Sjögren syndrome B antigen (SSB)/La were nearly twice as likely to develop an autoimmune disease as mothers with anti-SSA/Ro only. Only TGFβT/T was significantly higher in SLE mothers compared to asymptomatic mothers (p = 0.03). Conclusions: Continued follow-up of asymptomatic NL mothers is warranted since nearly half progress, albeit few develop SLE. While the anti-SSB/La antibodies may be a risk factor for progression, further work is needed to determine reliable biomarkers in otherwise healthy women with anti-SSA/Ro antibodies identified solely because of an NL child.Keywords
This publication has 39 references indexed in Scilit:
- Polymorphisms of TGF-beta1 in cystic fibrosis patientsClinical Immunology, 2006
- Maternal antibody responses to the 52‐kd SSA/RO p200 peptide and the development of fetal conduction defectsArthritis & Rheumatism, 2005
- Genetic association of cutaneous neonatal lupus with HLA class II and tumor necrosis factor α: Implications for pathogenesisArthritis & Rheumatism, 2004
- Genetic polymorphisms in tumor necrosis factor (TNF)-α and TNF-β in a population-based study of systemic lupus erythematosus: associations and interaction with the interleukin-1α-889 C/T polymorphismHuman Immunology, 2004
- Systemic lupus erythematosus and genetic variation in the interleukin 1 gene cluster: a population based study in the southeastern United StatesAnnals of the Rheumatic Diseases, 2004
- Long‐term followup of children with neonatal lupus and their unaffected siblingsArthritis & Rheumatism, 2002
- Classification criteria for Sjogren's syndrome: a revised version of the European criteria proposed by the American-European Consensus GroupAnnals of the Rheumatic Diseases, 2002
- Incomplete lupus erythematosus: results of a multicentre study under the supervision of the EULAR Standing Committee on International Clinical Studies Including Therapeutic Trials (ESCISIT)Rheumatology, 2001
- The 52‐kd protein as a target of intermolecular spreading of the immune response to components of the SS‐A/Ro‐SS‐B/La complexArthritis & Rheumatism, 1997
- Isolated congenital heart block. long‐term outcome of mothers and characterization of the immune response to ss‐a/ro and to ss‐b/laArthritis & Rheumatism, 1993