PMID- 18625627
OWN - NLM
STAT- MEDLINE
DCOM- 20090624
LR  - 20211020
IS  - 1468-2060 (Electronic)
IS  - 0003-4967 (Print)
IS  - 0003-4967 (Linking)
VI  - 68
IP  - 6
DP  - 2009 Jun
TI  - Disease progression in mothers of children enrolled in the Research Registry for 
      Neonatal Lupus.
PG  - 828-35
LID - 10.1136/ard.2008.088054 [doi]
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)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 Sjogren 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-Sjogren syndrome 
      A antigen (SSA/)Ro and anti-Sjogren 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.
FAU - Rivera, T L
AU  - Rivera TL
AD  - New York University School of Medicine, New York, 10016, USA. 
      tania.rivera@nyumc.org
FAU - Izmirly, P M
AU  - Izmirly PM
FAU - Birnbaum, B K
AU  - Birnbaum BK
FAU - Byrne, P
AU  - Byrne P
FAU - Brauth, J B
AU  - Brauth JB
FAU - Katholi, M
AU  - Katholi M
FAU - Kim, M Y
AU  - Kim MY
FAU - Fischer, J
AU  - Fischer J
FAU - Clancy, R M
AU  - Clancy RM
FAU - Buyon, J P
AU  - Buyon JP
LA  - eng
GR  - R01 AR042455/AR/NIAMS NIH HHS/United States
GR  - R01 AR42455-01/AR/NIAMS NIH HHS/United States
GR  - R37 AR042455/AR/NIAMS NIH HHS/United States
GR  - N01 AR042220/AR/NIAMS NIH HHS/United States
GR  - N01-AR-4-2220/AR/NIAMS NIH HHS/United States
PT  - Journal Article
PT  - Research Support, N.I.H., Extramural
PT  - Research Support, Non-U.S. Gov't
DEP - 20080714
PL  - England
TA  - Ann Rheum Dis
JT  - Annals of the rheumatic diseases
JID - 0372355
RN  - 0 (Antibodies, Antinuclear)
RN  - 0 (Autoantibodies)
RN  - 0 (SS-A antibodies)
RN  - 0 (SS-B antibodies)
RN  - 0 (Tumor Necrosis Factor-alpha)
SB  - IM
MH  - Antibodies, Antinuclear/immunology
MH  - Autoantibodies/blood
MH  - Disease Progression
MH  - Female
MH  - Follow-Up Studies
MH  - Genotype
MH  - Humans
MH  - Infant, Newborn
MH  - Lupus Erythematosus, Systemic/*congenital/genetics/immunology
MH  - *Mothers
MH  - Polymorphism, Genetic
MH  - Registries
MH  - Tumor Necrosis Factor-alpha/genetics
PMC - PMC3558032
MID - NIHMS433980
EDAT- 2008/07/16 09:00
MHDA- 2009/06/25 09:00
PMCR- 2013/01/29
CRDT- 2008/07/16 09:00
PHST- 2008/07/16 09:00 [pubmed]
PHST- 2009/06/25 09:00 [medline]
PHST- 2008/07/16 09:00 [entrez]
PHST- 2013/01/29 00:00 [pmc-release]
AID - ard.2008.088054 [pii]
AID - 10.1136/ard.2008.088054 [doi]
PST - ppublish
SO  - Ann Rheum Dis. 2009 Jun;68(6):828-35. doi: 10.1136/ard.2008.088054. Epub 2008 Jul 
      14.