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Disease progression in mothers of children enrolled in the Research Registry for Neonatal Lupus.
Ann Rheum Dis. 2009 Jun; 68(6):828-35.AR

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)alpha, 869T/C transforming growth factor (TGF)beta and -889C/T interleukin (IL)1alpha.

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 TGFbetaT/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.

Authors+Show Affiliations

New York University School of Medicine, New York, 10016, USA. tania.rivera@nyumc.orgNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article
Research Support, N.I.H., Extramural
Research Support, Non-U.S. Gov't

Language

eng

PubMed ID

18625627

Citation

Rivera, T L., et al. "Disease Progression in Mothers of Children Enrolled in the Research Registry for Neonatal Lupus." Annals of the Rheumatic Diseases, vol. 68, no. 6, 2009, pp. 828-35.
Rivera TL, Izmirly PM, Birnbaum BK, et al. Disease progression in mothers of children enrolled in the Research Registry for Neonatal Lupus. Ann Rheum Dis. 2009;68(6):828-35.
Rivera, T. L., Izmirly, P. M., Birnbaum, B. K., Byrne, P., Brauth, J. B., Katholi, M., Kim, M. Y., Fischer, J., Clancy, R. 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), 828-35. https://doi.org/10.1136/ard.2008.088054
Rivera TL, et al. Disease Progression in Mothers of Children Enrolled in the Research Registry for Neonatal Lupus. Ann Rheum Dis. 2009;68(6):828-35. PubMed PMID: 18625627.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR 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,M Y, AU - Fischer,J, AU - Clancy,R M, AU - Buyon,J P, Y1 - 2008/07/14/ PY - 2008/7/16/pubmed PY - 2009/6/25/medline PY - 2008/7/16/entrez SP - 828 EP - 35 JF - Annals of the rheumatic diseases JO - Ann. Rheum. Dis. VL - 68 IS - 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)alpha, 869T/C transforming growth factor (TGF)beta and -889C/T interleukin (IL)1alpha. 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 TGFbetaT/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. SN - 1468-2060 UR - https://www.unboundmedicine.com/medline/citation/18625627/Disease_progression_in_mothers_of_children_enrolled_in_the_Research_Registry_for_Neonatal_Lupus_ L2 - https://ard.bmj.com/cgi/pmidlookup?view=long&pmid=18625627 DB - PRIME DP - Unbound Medicine ER -