Disease progression in mothers of children enrolled in the Research Registry for Neonatal Lupus

T. L. Rivera, P. M. Izmirly, B. K. Birnbaum, P. Byrne, J. B. Brauth, M. Katholi, Mimi Kim, J. Fischer, R. M. Clancy, J. P. Buyon

Research output: Contribution to journalArticle

51 Citations (Scopus)

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 Sjö0gren 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.

Original languageEnglish (US)
Pages (from-to)828-835
Number of pages8
JournalAnnals of the Rheumatic Diseases
Volume68
Issue number6
DOIs
StatePublished - Jun 2009

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Registries
Disease Progression
Mothers
Lymphotoxin-beta
Research
Systemic Lupus Erythematosus
Antigens
Transforming Growth Factors
Biomarkers
Interleukin-1
Antibodies
Neonatal Systemic lupus erythematosus
Autoimmune Diseases
Parturition
SS-A antigen
Lupus Nephritis
Medical Records
SS-A antibodies

ASJC Scopus subject areas

  • Rheumatology
  • Immunology
  • Biochemistry, Genetics and Molecular Biology(all)
  • Immunology and Allergy

Cite this

Rivera, T. L., Izmirly, P. M., Birnbaum, B. K., Byrne, P., Brauth, J. B., Katholi, M., ... Buyon, J. P. (2009). Disease progression in mothers of children enrolled in the Research Registry for Neonatal Lupus. Annals of the Rheumatic Diseases, 68(6), 828-835. https://doi.org/10.1136/ard.2008.088054

Disease progression in mothers of children enrolled in the Research Registry for Neonatal Lupus. / Rivera, T. L.; Izmirly, P. M.; Birnbaum, B. K.; Byrne, P.; Brauth, J. B.; Katholi, M.; Kim, Mimi; Fischer, J.; Clancy, R. M.; Buyon, J. P.

In: Annals of the Rheumatic Diseases, Vol. 68, No. 6, 06.2009, p. 828-835.

Research output: Contribution to journalArticle

Rivera, TL, Izmirly, PM, Birnbaum, BK, Byrne, P, Brauth, JB, Katholi, M, Kim, M, Fischer, J, Clancy, RM & Buyon, JP 2009, 'Disease progression in mothers of children enrolled in the Research Registry for Neonatal Lupus', Annals of the Rheumatic Diseases, vol. 68, no. 6, pp. 828-835. https://doi.org/10.1136/ard.2008.088054
Rivera, T. L. ; Izmirly, P. M. ; Birnbaum, B. K. ; Byrne, P. ; Brauth, J. B. ; Katholi, M. ; Kim, Mimi ; Fischer, J. ; Clancy, R. M. ; Buyon, J. P. / Disease progression in mothers of children enrolled in the Research Registry for Neonatal Lupus. In: Annals of the Rheumatic Diseases. 2009 ; Vol. 68, No. 6. pp. 828-835.
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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 Sj{\"o}0gren 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-Sj{\"o}gren syndrome A antigen (SSA/)Ro and anti-Sj{\"o}gren 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.",
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T1 - Disease progression in mothers of children enrolled in the Research Registry for Neonatal Lupus

AU - Rivera, T. L.

AU - Izmirly, P. M.

AU - Birnbaum, B. K.

AU - Byrne, P.

AU - Brauth, J. B.

AU - Katholi, M.

AU - Kim, Mimi

AU - Fischer, J.

AU - Clancy, R. M.

AU - Buyon, J. P.

PY - 2009/6

Y1 - 2009/6

N2 - 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 Sjö0gren 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.

AB - 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 Sjö0gren 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.

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