新生儿红斑狼疮病例报告与文献复习
摘要
目的：为了提高临床医生对新生儿红斑狼疮的认识,进一步提高新生儿红斑狼疮的诊疗质量。
方法：对2例非临床红斑狼疮或干燥综合征患者所生的新生儿红斑狼疮患儿的临床病历资料进行分析总结,并结合相关文献讨论其发病病因、临床特点、组织病理学特征、诊断、鉴别诊断及治疗。
结果：根据2例新生儿的皮疹特点,患儿及生母ANA(±)、抗ENA：抗Ro/SSA(+).抗La/SSB(+),以及皮肤病理改变,并与其它有类似I临床表现的皮肤病进行鉴别,诊断新生儿红斑狼疮成立。
结论：新生儿狼疮临床上少见,非临床红斑狼疮或干燥综合征患者所生者更少见,容易误诊,但只要提高认识,根据典型的临床表现,结合母婴实验室检查,以及组织病理学改变,并经与相关疾病进行鉴别,可以确诊。
Objective:In order to improve medical doctors to understanding neonatal lupus erythematosus, and further knowledge of diagnosis and treatment of neonatal lupus.
Methods:Clinical data of two cases of neonatal lupus erythematosus born from mother who have no clinical features of lupus erythematosus and Sjogren syndrome were analyzed and related literatures associated with the causes, clinical manifestations, pathological changes, diagnosis and treatment were reviewed.
Results:Clinical manifestation of the patient is typical; laboratory tests of both mother and infant:ANA(+), antiRo/SSA(+), antiRo/SSB(+); histopatholo-gy changes are fixed with connective tissue disease and diagnose as neonatal lupus erythematosus after distinguished form other related diseases
Conclusion:Neonatal lupus erythematosus is rare, born by obvious clinical features of SLE and SS mother is more rare, easy to misdiagnose.But improve the knowledge of the NLE, can be maked exact diagnosis by characteristics of skin lesions, immuno-laboratory test of both mother and fetus and histopatho-logical changes after excluding the differential diagnosis.
方法：对2例非临床红斑狼疮或干燥综合征患者所生的新生儿红斑狼疮患儿的临床病历资料进行分析总结,并结合相关文献讨论其发病病因、临床特点、组织病理学特征、诊断、鉴别诊断及治疗。
结果：根据2例新生儿的皮疹特点,患儿及生母ANA(±)、抗ENA：抗Ro/SSA(+).抗La/SSB(+),以及皮肤病理改变,并与其它有类似I临床表现的皮肤病进行鉴别,诊断新生儿红斑狼疮成立。
结论：新生儿狼疮临床上少见,非临床红斑狼疮或干燥综合征患者所生者更少见,容易误诊,但只要提高认识,根据典型的临床表现,结合母婴实验室检查,以及组织病理学改变,并经与相关疾病进行鉴别,可以确诊。
Objective:In order to improve medical doctors to understanding neonatal lupus erythematosus, and further knowledge of diagnosis and treatment of neonatal lupus.
Methods:Clinical data of two cases of neonatal lupus erythematosus born from mother who have no clinical features of lupus erythematosus and Sjogren syndrome were analyzed and related literatures associated with the causes, clinical manifestations, pathological changes, diagnosis and treatment were reviewed.
Results:Clinical manifestation of the patient is typical; laboratory tests of both mother and infant:ANA(+), antiRo/SSA(+), antiRo/SSB(+); histopatholo-gy changes are fixed with connective tissue disease and diagnose as neonatal lupus erythematosus after distinguished form other related diseases
Conclusion:Neonatal lupus erythematosus is rare, born by obvious clinical features of SLE and SS mother is more rare, easy to misdiagnose.But improve the knowledge of the NLE, can be maked exact diagnosis by characteristics of skin lesions, immuno-laboratory test of both mother and fetus and histopatho-logical changes after excluding the differential diagnosis.
引文
1. McCuistion CH, Schoch EP. Possible discoid lupus erythematosus in new born infant. Arch Dermatol Syph,1954;70:782-784.
2. Wei S, Tian MY, Li HC, et al. Neonatal Lupus Erythematosus:Three case Reports and Review of the Chinese Literature. Clinical Pediatrics,2010; 49 (7):627-634.
3. Reed BR, Lee LA, Harmon C, et al. Autoantibodies to SSA/Ro in infants with congenital heart block. J Pediatric,1983; 103(6):889-891.
4. Clancy RM, Askanase AD, Kapur RP, et al. Transdifferentiation of a cardiac fibroblasts, a fetal factor in anti-SSA/Ro-SSB/La antibody mediated congenital heart block. J Immunol,2002; 169(4):2156-2163.
5. Eftekhari P, Salle L, Lezoualch F, et al. Anti-SSA/Ro52 autoantibodies blocking the cardiac 5-HT4 serotoninergic receptor could explain neonatal lupus congenital heart block. EurJ Immunol,2000; 30(10):2782-2790.
6. Boh EE. Neonatal lupus erythematosus. Clinical Dermatology,2004; 22: 125-128.
7. Ritsuko K, Sumiyuki M, Toshiaki N, et al. Neonatal lupus erythematosus in Japan:A review of the literature. Autoimmunity Reviews,2009; 462-466.
8. Petri M, Watson R, Hochberg MC. Anti-Ro antibodies and neonatal lupus. Rheum Dis Clin North Am,1989; 15(2):335-360.
9. James TC, Ross EP. Textbook of Peditric Rheumatology, Philadelphial. Saunders Company,1995; 301-302.
10. Penate Y, Lujan D, Rodriguez J, et al. Neonatal lupus erythematosus:4 cases and clinical review. Actas Dermosifiliogr,2005; 96(10):690-696.
11. Asboth D, Kassay E, Noll J, et al. Neonatal lupus erythematosus:deep and ulcerating form. Borgyogaszati Venerol Szemle,2000; 76:263-265.
12. Lynn CC, Galbraith S, Holland K. Congenital lupus erythematosus present-ing at birth with widespread erosions, pancytopenia and subsequent hepato-biliary disease. Pediatric Dermatology,2010; 27(1):109-111.
13. Cimaz R, Biggioggero M, Catelli L, et al. Ultraviolet light exposure is not a requirement for the development of cutaneous neonatal lupus. Lupus,2002; 11(4):257-260.
14. Wisuthsarewong W, Soongswang J, Chantorn R. Neonatal lupus erythemat-osus:clinical character, investigation, and outcome.Pediatr Dermatol,2011; 28(2):115-121.
15. 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(8):1832-1835.
16.凤岐,王江,周怡芳,张立新。新生儿狼疮综合征八例报告并文献复习。中华风湿病学杂志,2004；8(10)：611-614.
17. Friedman DM, Llanos C, Izmirly PM, et al. Evaluation of fetuses in a study of intravenous immunoglobulin as preventive therapy for congenital heart block:Results of a multicenter, prospective, open label clinical trial.Arthritis Rheum,2010; 62(4):1138-1146.
18. Pisoni CN, Brucato A, Ruffatti A, et al. Failure of intravenous immuneglo-bulin to prevent congenital heart block:Findings of a multicenter, prospecti-ve, observational study. Arthritis Rheum,2010; 62(4):1147-1152.
19. Izmirly PM, Kim MY, Llanos C, et al. Evaluation of the risk of anti-SSA/ Ro-SSB/La antibody-associated cardiac manifestations of neonatal lupus in fetuses of mothers with systemic lupus erythematosus exposed to hydroxyl-chloroquine. Ann Rheum Dis,2010;69(10):1827-1830.
20.赵辨。中国临床皮肤病学。南京：江苏科学技术出版社,2010：398-399.
21. 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(9):2377-2383.
22. Guettrot IG, Cohen L, Fermont L, et al. A new presentation of neonatal lupus:5 cases of isolated mild endocardial fibroelastosis associated with maternal Anti-SSA/Ro and Anti-SSB/La antibodies. J Rheumatol,2011; 38(2):378-386.
1. 赵辨.中国临床皮肤病学.南京：江苏科学技术出版社,2010;398-399.
2. Wei S, Tian MY, Li HC, et al. Neonatal lupus erythematosus:Three case reports and review of the Chinese literature. Clinical Pediatrics,2010; 49(7): 627-634.
3. McCuistion CH, Schoch EP. Possible discoid lupus erythematosus in newborn infant. Arch Dermatol Syph,1954;70:782-784.
4. Cai ZW, Ji YY, Yao GB. A case of neonatal lupus erythematosus in chinese. Shanghai J Immunol,1991; 11:363.
5. Reed BR, Lee LA, Harmon C, et al. Autoantibodies to SSA/Ro in infants with congenital heart block. J Pediatric,1983; 103(6):889-891.
6. Clancy RM, Askanase AD, Kapur RP, et al. Transdifferentiation of a cardiac fibroblasts, a fetal factor in antiSSA/Ro, SSB/La antibody mediated congenital heart block J Immunol,2002; 169(4):2156-2163.
7. Eftekhari P, Salle L, Lezoualch F, et al. Anti-SSA/Ro52 autoantibodies blocking the cardiac 5-HT4 serotoninergic receptor could explain neonatal lupus congenital heart block. EurJ Immunol,2000; 30(10):2782-2790.
8. Boh EE. Neonatal lupus erythematosus. Clinical Dermatology,2004; 22: 125-128.
9. Wisuthsarewong W, Soongswang J, Chantorn R. Neonatal lupus erythe-matosus:clinical character, investigation, and outcome. Pediatr Dermatol, 2011; 28(2):115-121.
10. Penate Y, Lujan D, Rodriguez J, et al. Neonatal lupus erythematosus:4 cases and clinical review. Actas Dermosifiliogr,2005; 96(10):690-696.
11. Asboth D, Kassay E, Noll J, et al. Neonatal lupus erythematosus:deep and ulcerating form. Borgyogaszati Venerol Szemle,2000; 76:263-265.
12. Lynn CC, Galbraith S, Holland K. Congenital lupus erythematosus present-ing at birth with widespread erosions, pancytopenia and subsequent hepato-biliary disease. Pediatr Dermatol,2010; 27(1):109-111.
13. Cimaz R, Biggioggero M, Catelli L, et al. Ultraviolet light exposure is not a requirement for the development of cutaneous neonatal lupus. Lupus,2002; 11(4):257-260.
14. Kimiko N, Hiroshi Wa, Hajime K, et al. Neonatal lupus erythematosus in identical twins, showing transient bullous lesions. Pediatric Dermatology, 2011:28(4):397-400.
15. 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(8):1832-1835.
16. Guettrot IG, Cohen L, Fermont L, et al. A new presentation of neonatal lupus:5 cases of isolated mild endocardial fibroelastosis associated with maternal Anti-SSA/Ro and Anti-SSB/La antibodies. J Rheumatol,2011; 38(2):378-386.
17. Prendiville JS, Cabral DA, Poskitt KJ,et al. Central nervous system invol-vement in neonatal lupus erythematosus. Pediatric dermatology,2003; 20 (1):60-67.
18. Spalding SJ, Hennon T, Dohar J,et al.Neonatal lupus erythematosus comp-licated by mucocutaneous and visceral hemangiomas. Lupus,2007; 16:904-907.
19. Spence D, Hornberger L, Hamilton R,et al. Increased risk of complete congenital heart block in infants born to women with hypothyroidism and anti-Ro and/or anti-La antibodies. J Rheumatol,2006; 33(1):167-170.
20. McKinlay JR, Cooke LM, Cunningham BB, et al. Neonatal lupus erythem-atosus. J Am Board Fam Pract,2001;14(1):68-70.
21.凤岐,王江,周怡芳,张立新。新生儿狼疮综合征八例报告并文献复习。中华风湿病学杂志,2004；8(10)：611-614.
22. Izmirly PM, Saxena A, Kim MY, et al. Maternal and fetal factors associate-ed with mortality and morbidity in a multi-racial/ethnic registry of anti-SSA/Ro-associated cardiac neonatal lupus. Rheum Dis Clin North Am, 2007; 33(2):267-285
23. Friedman DM, Llanos C, Izmirly PM, et al. Evaluation of fetuses in a study of intravenous immunoglobulin as preventive therapy for congenital heart block:Result of a multicenter,prospective, open label clinical trial. Arthritis Rheum,2010; 62(4):1138-1146.
24. Pisoni CN, Brucato A, Ruffatti A, et al. Failure of intravenous immunoglo-bulin to prevent congenital heart block:Findings of a multicenter, prospect-ive, observational study. Arthritis Rheum,2010; 62(4):1147-1152.
25. Izmirly PM, Kim MY, Llanos C,et al. Evaluation of the risk of antiSSA/ Ro-SSB/La antibody-associated cardiac manifestations of neonatal lupus in fetuses of mothers with systemic lupus erythematosus exposed to hydroxyl-chloroquine. Ann Rheum Dis,2010; 69(10):1827-1830.
26. Yang CH, Chen JY, Lee SC, et al. Successful preventive treatment of cong-enital heart block during pregnancy in a woman with systemic lupus erythe-matosus and anti-Sjogren's syndrome A/Ro antibody. J Microbiol Immunol Infect,2005; 38(5):365-369.
27. 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(9):2377-2383.
2. Wei S, Tian MY, Li HC, et al. Neonatal Lupus Erythematosus:Three case Reports and Review of the Chinese Literature. Clinical Pediatrics,2010; 49 (7):627-634.
3. Reed BR, Lee LA, Harmon C, et al. Autoantibodies to SSA/Ro in infants with congenital heart block. J Pediatric,1983; 103(6):889-891.
4. Clancy RM, Askanase AD, Kapur RP, et al. Transdifferentiation of a cardiac fibroblasts, a fetal factor in anti-SSA/Ro-SSB/La antibody mediated congenital heart block. J Immunol,2002; 169(4):2156-2163.
5. Eftekhari P, Salle L, Lezoualch F, et al. Anti-SSA/Ro52 autoantibodies blocking the cardiac 5-HT4 serotoninergic receptor could explain neonatal lupus congenital heart block. EurJ Immunol,2000; 30(10):2782-2790.
6. Boh EE. Neonatal lupus erythematosus. Clinical Dermatology,2004; 22: 125-128.
7. Ritsuko K, Sumiyuki M, Toshiaki N, et al. Neonatal lupus erythematosus in Japan:A review of the literature. Autoimmunity Reviews,2009; 462-466.
8. Petri M, Watson R, Hochberg MC. Anti-Ro antibodies and neonatal lupus. Rheum Dis Clin North Am,1989; 15(2):335-360.
9. James TC, Ross EP. Textbook of Peditric Rheumatology, Philadelphial. Saunders Company,1995; 301-302.
10. Penate Y, Lujan D, Rodriguez J, et al. Neonatal lupus erythematosus:4 cases and clinical review. Actas Dermosifiliogr,2005; 96(10):690-696.
11. Asboth D, Kassay E, Noll J, et al. Neonatal lupus erythematosus:deep and ulcerating form. Borgyogaszati Venerol Szemle,2000; 76:263-265.
12. Lynn CC, Galbraith S, Holland K. Congenital lupus erythematosus present-ing at birth with widespread erosions, pancytopenia and subsequent hepato-biliary disease. Pediatric Dermatology,2010; 27(1):109-111.
13. Cimaz R, Biggioggero M, Catelli L, et al. Ultraviolet light exposure is not a requirement for the development of cutaneous neonatal lupus. Lupus,2002; 11(4):257-260.
14. Wisuthsarewong W, Soongswang J, Chantorn R. Neonatal lupus erythemat-osus:clinical character, investigation, and outcome.Pediatr Dermatol,2011; 28(2):115-121.
15. 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(8):1832-1835.
16.凤岐,王江,周怡芳,张立新。新生儿狼疮综合征八例报告并文献复习。中华风湿病学杂志,2004；8(10)：611-614.
17. Friedman DM, Llanos C, Izmirly PM, et al. Evaluation of fetuses in a study of intravenous immunoglobulin as preventive therapy for congenital heart block:Results of a multicenter, prospective, open label clinical trial.Arthritis Rheum,2010; 62(4):1138-1146.
18. Pisoni CN, Brucato A, Ruffatti A, et al. Failure of intravenous immuneglo-bulin to prevent congenital heart block:Findings of a multicenter, prospecti-ve, observational study. Arthritis Rheum,2010; 62(4):1147-1152.
19. Izmirly PM, Kim MY, Llanos C, et al. Evaluation of the risk of anti-SSA/ Ro-SSB/La antibody-associated cardiac manifestations of neonatal lupus in fetuses of mothers with systemic lupus erythematosus exposed to hydroxyl-chloroquine. Ann Rheum Dis,2010;69(10):1827-1830.
20.赵辨。中国临床皮肤病学。南京：江苏科学技术出版社,2010：398-399.
21. 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(9):2377-2383.
22. Guettrot IG, Cohen L, Fermont L, et al. A new presentation of neonatal lupus:5 cases of isolated mild endocardial fibroelastosis associated with maternal Anti-SSA/Ro and Anti-SSB/La antibodies. J Rheumatol,2011; 38(2):378-386.
1. 赵辨.中国临床皮肤病学.南京：江苏科学技术出版社,2010;398-399.
2. Wei S, Tian MY, Li HC, et al. Neonatal lupus erythematosus:Three case reports and review of the Chinese literature. Clinical Pediatrics,2010; 49(7): 627-634.
3. McCuistion CH, Schoch EP. Possible discoid lupus erythematosus in newborn infant. Arch Dermatol Syph,1954;70:782-784.
4. Cai ZW, Ji YY, Yao GB. A case of neonatal lupus erythematosus in chinese. Shanghai J Immunol,1991; 11:363.
5. Reed BR, Lee LA, Harmon C, et al. Autoantibodies to SSA/Ro in infants with congenital heart block. J Pediatric,1983; 103(6):889-891.
6. Clancy RM, Askanase AD, Kapur RP, et al. Transdifferentiation of a cardiac fibroblasts, a fetal factor in antiSSA/Ro, SSB/La antibody mediated congenital heart block J Immunol,2002; 169(4):2156-2163.
7. Eftekhari P, Salle L, Lezoualch F, et al. Anti-SSA/Ro52 autoantibodies blocking the cardiac 5-HT4 serotoninergic receptor could explain neonatal lupus congenital heart block. EurJ Immunol,2000; 30(10):2782-2790.
8. Boh EE. Neonatal lupus erythematosus. Clinical Dermatology,2004; 22: 125-128.
9. Wisuthsarewong W, Soongswang J, Chantorn R. Neonatal lupus erythe-matosus:clinical character, investigation, and outcome. Pediatr Dermatol, 2011; 28(2):115-121.
10. Penate Y, Lujan D, Rodriguez J, et al. Neonatal lupus erythematosus:4 cases and clinical review. Actas Dermosifiliogr,2005; 96(10):690-696.
11. Asboth D, Kassay E, Noll J, et al. Neonatal lupus erythematosus:deep and ulcerating form. Borgyogaszati Venerol Szemle,2000; 76:263-265.
12. Lynn CC, Galbraith S, Holland K. Congenital lupus erythematosus present-ing at birth with widespread erosions, pancytopenia and subsequent hepato-biliary disease. Pediatr Dermatol,2010; 27(1):109-111.
13. Cimaz R, Biggioggero M, Catelli L, et al. Ultraviolet light exposure is not a requirement for the development of cutaneous neonatal lupus. Lupus,2002; 11(4):257-260.
14. Kimiko N, Hiroshi Wa, Hajime K, et al. Neonatal lupus erythematosus in identical twins, showing transient bullous lesions. Pediatric Dermatology, 2011:28(4):397-400.
15. 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(8):1832-1835.
16. Guettrot IG, Cohen L, Fermont L, et al. A new presentation of neonatal lupus:5 cases of isolated mild endocardial fibroelastosis associated with maternal Anti-SSA/Ro and Anti-SSB/La antibodies. J Rheumatol,2011; 38(2):378-386.
17. Prendiville JS, Cabral DA, Poskitt KJ,et al. Central nervous system invol-vement in neonatal lupus erythematosus. Pediatric dermatology,2003; 20 (1):60-67.
18. Spalding SJ, Hennon T, Dohar J,et al.Neonatal lupus erythematosus comp-licated by mucocutaneous and visceral hemangiomas. Lupus,2007; 16:904-907.
19. Spence D, Hornberger L, Hamilton R,et al. Increased risk of complete congenital heart block in infants born to women with hypothyroidism and anti-Ro and/or anti-La antibodies. J Rheumatol,2006; 33(1):167-170.
20. McKinlay JR, Cooke LM, Cunningham BB, et al. Neonatal lupus erythem-atosus. J Am Board Fam Pract,2001;14(1):68-70.
21.凤岐,王江,周怡芳,张立新。新生儿狼疮综合征八例报告并文献复习。中华风湿病学杂志,2004；8(10)：611-614.
22. Izmirly PM, Saxena A, Kim MY, et al. Maternal and fetal factors associate-ed with mortality and morbidity in a multi-racial/ethnic registry of anti-SSA/Ro-associated cardiac neonatal lupus. Rheum Dis Clin North Am, 2007; 33(2):267-285
23. Friedman DM, Llanos C, Izmirly PM, et al. Evaluation of fetuses in a study of intravenous immunoglobulin as preventive therapy for congenital heart block:Result of a multicenter,prospective, open label clinical trial. Arthritis Rheum,2010; 62(4):1138-1146.
24. Pisoni CN, Brucato A, Ruffatti A, et al. Failure of intravenous immunoglo-bulin to prevent congenital heart block:Findings of a multicenter, prospect-ive, observational study. Arthritis Rheum,2010; 62(4):1147-1152.
25. Izmirly PM, Kim MY, Llanos C,et al. Evaluation of the risk of antiSSA/ Ro-SSB/La antibody-associated cardiac manifestations of neonatal lupus in fetuses of mothers with systemic lupus erythematosus exposed to hydroxyl-chloroquine. Ann Rheum Dis,2010; 69(10):1827-1830.
26. Yang CH, Chen JY, Lee SC, et al. Successful preventive treatment of cong-enital heart block during pregnancy in a woman with systemic lupus erythe-matosus and anti-Sjogren's syndrome A/Ro antibody. J Microbiol Immunol Infect,2005; 38(5):365-369.
27. 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(9):2377-2383.