ORIGINAL PAPER
Infants of mothers with systemic connective tissue disease – serological and clinical status in the first year of life; a pilot study
1
Department of Neonatology, Pomeranian Medical University, Szczecin, Poland
2
Independent Laboratory for Rheumatologic Diagnostics, Pomeranian Medical University in Szczecin, Poland
3
Department of Rheumatology Pomeranian Medical University, Szczecin, Poland
Submission date: 2020-12-22
Final revision date: 2021-01-30
Acceptance date: 2021-01-31
Publication date: 2021-03-28
Pediatr Pol 2021;96(1):31-37
KEYWORDS
TOPICS
ABSTRACT
Introduction:
Neonatal lupus erythematosus (NLE) is an uncommon autoimmune disease caused by transplacental passage of maternal antibodies. The aim of the study was to investigate whether antinuclear antibodies (ANA) detected in newborns of mothers with systemic connective tissue disease (SCTD) are present after 6 months of age, and whether they are accompanied by clinical symptoms, the so-called NLE.
Material and methods:
A prospective pilot study was conducted in a group of 20 full-term newborns (11 males and 9 females) consequently born to mothers with SCTD and 6 newborns (3 males and 3 females) of mothers without autoimmune disease. The infants were examined for symptoms of NLE. The ANA profile was evaluated in newborns and repeated at the age of 7 months by using the indirect immunofluorescence technique.
Results:
ANA was detected in 16/20 newborns from the study group in titre 1 : 40 to 1 : 2560 and in 4/6 newborns from the control group in titre 1 : 40 to 1 : 320, as well as in 9 of 19 7-months-old babies of SCTD mothers. There were no skin lesions, and elevated liver enzymes were found in 8 newborns and haematological changes in 7 newborns. No pathological changes were found in the ECG examination in the neonatal period in any of the children.
Conclusions:
Maternal ANAs are detected in newborns and in 7-month-old babies. Despite 80% of ANA detection in the neonatal period of infants of SCTD mothers and 47% detection in 7-month-old babies, there were no evident symptoms of NLE besides some elevated levels of liver enzymes and haematological changes.
Neonatal lupus erythematosus (NLE) is an uncommon autoimmune disease caused by transplacental passage of maternal antibodies. The aim of the study was to investigate whether antinuclear antibodies (ANA) detected in newborns of mothers with systemic connective tissue disease (SCTD) are present after 6 months of age, and whether they are accompanied by clinical symptoms, the so-called NLE.
Material and methods:
A prospective pilot study was conducted in a group of 20 full-term newborns (11 males and 9 females) consequently born to mothers with SCTD and 6 newborns (3 males and 3 females) of mothers without autoimmune disease. The infants were examined for symptoms of NLE. The ANA profile was evaluated in newborns and repeated at the age of 7 months by using the indirect immunofluorescence technique.
Results:
ANA was detected in 16/20 newborns from the study group in titre 1 : 40 to 1 : 2560 and in 4/6 newborns from the control group in titre 1 : 40 to 1 : 320, as well as in 9 of 19 7-months-old babies of SCTD mothers. There were no skin lesions, and elevated liver enzymes were found in 8 newborns and haematological changes in 7 newborns. No pathological changes were found in the ECG examination in the neonatal period in any of the children.
Conclusions:
Maternal ANAs are detected in newborns and in 7-month-old babies. Despite 80% of ANA detection in the neonatal period of infants of SCTD mothers and 47% detection in 7-month-old babies, there were no evident symptoms of NLE besides some elevated levels of liver enzymes and haematological changes.
REFERENCES (37)
1.
Arroyava CM, Giambrone MJ, Rich KC, Walaszek M. The frequency of antinuclear antibody (ANA) in children by use of mouse kidney (MK) and human epithelial cells (HEp-2) as substrates. J Allergy Clin Immunol 1988; 82: 741-744.
2.
Tan EM, Feltkamp TEW, Smolen JS, et al. Range of antinuclear antibodies in “healthy” individuals. Arthritis Rheum 1997; 40: 1601-1611.
3.
Li X, Liu X, Cui J, et al. Epidemiological survey of antinuclear antibodies in healthy population and analysis of clinical characteristics of positive population. J Clin Lab Anal 2019; 33: e22965.
4.
Cervera R, Font J, Ramos-Casals M, et al. Primary Sjögren’s syndrome in men: clinical and immunological characteristics. Lupus 2000; 9: 61-64.
5.
Barnett AJ, McNeilage LJ. Antinuclear antibodies in patients with scleroderma (systemic sclerosis) and in their blood relatives and spouses. Ann Rheum Dis 1993; 52: 365-368.
6.
Burdt MA, Hoffman RW, Deutscher S, et al. Long-term outcome in mixed connective tissue disease: Longitudinal clinical and serologic findings. Arthritis Rheum 1999; 42: 899-909.
7.
Obermayer-Straub P, Strassburg CP, Manns MP. Autoimmune hepatitis. J Hepatol 2000; 32 (1 Suppl): 181-197.
8.
Kavanaugh A, Tomar R, Reveille J, et al.Guidelines for clinical use of the antinuclear antibody test and tests for specific autoantibodies to nuclear antigens. American College of Pathologists. Arch Pathol Lab Med 2000; 124: 71-81.
9.
Kumar Y, Bhatia A, Minz RW. Antinuclear antibodies and their detection methods in diagnosis of connective tissue diseases: a journey revisited. Diagn Pathol 2009; 4: 1-10.
10.
Hon KL, Alexander KC, Leung AKC. Neonatal lupus erythematosus. Autoimmune Dis 2012; 2012: 301274.
11.
Yang Q, Shao X-M, Cao Y, et al. Neonatal lupus erythematosus: analysis of 8 cases. Zhonghua Er Ke Za Zhi 2008; 46: 18-21.
12.
Lee LA. Transient autoimmunity related to maternal autoantibodies: neonatal lupus. Autoimmun Rev 2005; 4: 207-213.
13.
Herrera-Esparza R, Bollain-y-Goytia JJ, Avalos-Díaz E. Pathogenic effects of maternal antinuclear antibodies during pregnancy in women with lupus. Rheum Rep 2014; 6: 5545.
14.
Shahian M, Khosravi A, Anbardar MH. Early cholestasis in neonatal lupus erythematosus. Ann Saudi Med 2011; 31: 80-82.
15.
Buyon JP. Neonatal Lupus: bedside to bench and back. Scand Rheumatol 1996; 25: 271-276.
16.
Li L, Dong GF, Han FZ, et al. Neonatal lupus erythematosus: a report of 7 cases and review of 87 cases of China. Zhonghua Er Ke Za Zhi 2011; 49: 146-150.
17.
Liu J, Yang YH, Lin YT, Chiang BL. Clinical characteristics of neonatal lupus erythematosus. J Microbiol Immunol Infect 2001; 34: 265-268.
19.
Wisuthsarewong W, Soongswang J, Chantorn R. Neonatal lupus erythematosus: clinical character, investigation, and outcome. Pediat Dermat 2011; 28: 115-121.
20.
Jaeggi E, Laskin C, Hamilton R, et al. The importance of the level of maternal anti-Ro/SSA antibodies as a prognostic marker of the development of cardiac neonatal lupus erythematosus. A Prospective Study of 186 antibody-exposed fetuses and infants. J Am Coll Cardiol 2010; 55: 2778-2784.
21.
Inzinger M, Salmhofer W, Binder B. Neonatal Lupus Erythematosus and its clinical variability. J Dtsch Dermatol Ges 2012; 10: 407-411.
22.
Zuppa AA, Fracchiolla A, Cota F, et al. Infants born to mothers with anti-SSA/Ro autoantibodies: neonatal outcome and follow-up. Clin Pediatr 2008; 47: 231-236.
23.
Diaz-Frias J, Badri T. Neonatal Lupus Erythematosus. In: StatPearls Treasure. StatPearls Publishing LLC, Island 2020.
24.
Kociszewska-Najman B, Jaskólska M, Joanna Schreiber-Zamora J, et al. Toczeń rumieniowaty noworodków. Stanowisko Zespołu Ekspertów w sprawie diagnostyki, monitorowania oraz leczenia matki i noworodka. Gin Perinat Prakt 2020; 1: 33-43.
25.
Torres-Navarro I, López-Montesinos B, Évole-Buselli M. Neonatal lupus. An Pediatr (Barc) 2021; 94: 122-123.
26.
Izmirly PM, Llanos C, Lee LA, et al. Cutaneous manifestations of neonatal lupus and risk of subsequent congenital heart block. Arthritis Rheum 2010; 62: 1153-1157.
27.
Brucato A, Frassi M, Franceschini F, et al. Risk of congenital complete heart block in newborns of mothers with anti-Ro/SSA antibodies detected by counterimmunoelectrophoresis: a prospective study of 100 women. Arthritis Rheum 2001; 44: 1832-1835.
28.
Zuppa AA, Riccardi R, Frezza S. Neonatal lupus: Follow-up in infants with anti-SSA/Ro antibodies and review of the literature. Autoimmun Rev 2017; 16: 427-432.
29.
Motta M, Chirico G, Biasini-Rebaioli Ch, et al. Anticardiolipin and anti-beta2 glycoprotein I antibodies in infants born to mothers with antiphospholipid antibody-positive autoimmune disease: a follow-up study. Am J Perinatol 2006; 23: 247-251.
30.
Sanchez-Manubens J, Ortiz-Santamaria V, Coll Sibina MT, et al. Newborns whose mother has autoimmune disease. A community hospitals’ experience. Reumatol Clin 2013; 9: 161-165.
31.
Beck JS, Oakley CL, Rowell NR. Transplacental passage of antinuclear antibody study in infants of mothers with systemic lupus erythematosus. Arch Dermatol 1966; 93: 656-663.
32.
Silverman E, Jaeggi E. Non-cardiac manifestations of neonatal lupus erythematosus. Scan J Immunol 2010; 72: 223-225.
33.
Sun W, Yuan TM, Chen LH, Yu HM. Neonatal lupus erythematosus: three case reports and review of the chinese literature. Clin Pediatr 2010; 49: 627-634.
34.
Savino F, Viola S, Tarasco V, et al. Neonatal lupus erythematosus: a cutaneous cases based update. Ital J Pediatr 2016; 42: 1.
35.
Neiman A, Lee L, Weston W, Buyon J. Cutaneous manifestations of neonatal lupus without heart block: characteristics of mothers and children enrolled in a national registry. J Pediatr 2000; 137: 674-680.
36.
Singalavanija S, Limpongsanurak W, Aoongern S. Neonatal lupus erythematosus: a 20-year retrospective study. J Med Assoc Thai 2014; 97 Suppl 6: S74-S82.
37.
Martin V, Lee LA, Askanase AD, et al. Long-term follow-up of children with neonatal lupus and their unaffected siblings. Arthritis Rheum 2002; 46: 2377-2383.
Share
RELATED ARTICLE
We process personal data collected when visiting the website. The function of obtaining information about users and their behavior is carried out by voluntarily entered information in forms and saving cookies in end devices. Data, including cookies, are used to provide services, improve the user experience and to analyze the traffic in accordance with the Privacy policy. Data are also collected and processed by Google Analytics tool (more).
You can change cookies settings in your browser. Restricted use of cookies in the browser configuration may affect some functionalities of the website.
You can change cookies settings in your browser. Restricted use of cookies in the browser configuration may affect some functionalities of the website.
