Neonatal Lupus Erythematosus: A Case Report and Review of the Literature
Main Article Content
Keywords
neonatal lupus erythematosus, systemic lupus erythematosus, congenital heart block
Abstract
Neonatal lupus erythematosus (NLE) is a rare autoimmune condition caused by transplacental passage of maternal IgG autoantibodies to the fetus. It often presents with erythematous annular lesions on the scalp and periorbital area that appear after exposure to sunlight. Clinicians should be aware of the broad manifestations of neonatal lupus erythematosus, including cutaneous, cardiac, hematologic, hepatic and neurological manifestations. We present a case of cutaneous neonatal lupus erythematosus with rare central nervous system findings and perform a literature review on the subject.
References
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2. Chen CC, Lin KL, Chen CL, Wong AM, Huang JL. Central nervous system manifestations of neonatal lupus: a systematic review. Lupus. Dec 2013;22(14):1484-8. doi:10.1177/0961203313509294
3. 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. Jun 2009;68(6):828-35. doi:10.1136/ard.2008.088054
4. Shahian M, Khosravi A, Anbardar MH. Early cholestasis in neonatal lupus erythematosus. Ann Saudi Med. Jan-Feb 2011;31(1):80-2. doi:10.4103/0256-4947.70569
5. Boros CA, Spence D, Blaser S, Silverman ED. Hydrocephalus and macrocephaly: new manifestations of neonatal lupus erythematosus. Arthritis Rheum. Mar 15 2007;57(2):261-6. doi:10.1002/art.22543
6. Lee LA. Transient autoimmunity related to maternal autoantibodies: neonatal lupus. Autoimmun Rev. Apr 2005;4(4):207-13. doi:10.1016/j.autrev.2004.11.003
7. Izmirly PM, Saxena A, Sahl SK, et al. Assessment of fluorinated steroids to avert progression and mortality in anti-SSA/Ro-associated cardiac injury limited to the fetal conduction system. Ann Rheum Dis. Jun 2016;75(6):1161-5. doi:10.1136/annrheumdis-2015-208311
8. Izmirly PM, Saxena A, Kim MY, et al. Maternal and fetal factors associated with mortality and morbidity in a multi-racial/ethnic registry of anti-SSA/Ro-associated cardiac neonatal lupus. Circulation. Nov 1 2011;124(18):1927-35. doi:10.1161/circulationaha.111.033894
9. Cimaz R, Spence DL, Hornberger L, Silverman ED. Incidence and spectrum of neonatal lupus erythematosus: a prospective study of infants born to mothers with anti-Ro autoantibodies. J Pediatr. Jun 2003;142(6):678-83. doi:10.1067/mpd.2003.233
10. Vanoni F, Lava SAG, Fossali EF, et al. Neonatal Systemic Lupus Erythematosus Syndrome: a Comprehensive Review. Clin Rev Allergy Immunol. Dec 2017;53(3):469-476. doi:10.1007/s12016-017-8653-0
11. Strandberg L, Salomonsson S, Bremme K, Sonesson S, Wahren-Herlenius M. Ro52, Ro60 and La IgG autoantibody levels and Ro52 IgG subclass profiles longitudinally throughout pregnancy in congenital heart block risk pregnancies. Lupus. 2006;15(6):346-53. doi:10.1191/0961203306lu2309oa
12. Yoshimi R, Ueda A, Ozato K, Ishigatsubo Y. Clinical and pathological roles of Ro/SSA autoantibody system. Clin Dev Immunol. 2012;2012:606195. doi:10.1155/2012/606195
13. Gleicher N, Elkayam U. Preventing congenital neonatal heart block in offspring of mothers with anti-SSA/Ro and SSB/La antibodies: a review of published literature and registered clinical trials. Autoimmun Rev. Sep 2013;12(11):1039-45. doi:10.1016/j.autrev.2013.04.006
14. Provost TT, Watson R, Gammon WR, Radowsky M, Harley JB, Reichlin M. The neonatal lupus syndrome associated with U1RNP (nRNP) antibodies. N Engl J Med. Apr 30 1987;316(18):1135-8. doi:10.1056/nejm198704303161807
15. Sheth AP, Esterly NB, Ratoosh SL, Smith JP, Hebert AA, Silverman E. U1RNP positive neonatal lupus erythematosus: association with anti-La antibodies? Br J Dermatol. Apr 1995;132(4):520-6. doi:10.1111/j.1365-2133.1995.tb08705.x
16. Shiiya C, Ota M. Facial Rash, Fever, and Anemia in a Newborn. Jama. May 23 2017;317(20):2125-2126. doi:10.1001/jama.2017.3058
17. Lee LA. Neonatal lupus: clinical features and management. Paediatr Drugs. 2004;6(2):71-8. doi:10.2165/00148581-200406020-00001
18. Sommer LL, Manders SM. Seeing double: annular diaper rash in twins. Pediatr Dermatol. May-Jun 2015;32(3):410-3. doi:10.1111/pde.12508
19. See A, Wargon O, Lim A, Sugo E, Le K. Neonatal lupus erythematosus presenting as papules on the feet. Australas J Dermatol. Aug 2005;46(3):172-6. doi:10.1111/j.1440-0960.2005.00173.x
20. Silverman E, Jaeggi E. Non-cardiac manifestations of neonatal lupus erythematosus. Scand J Immunol. Sep 2010;72(3):223-5. doi:10.1111/j.1365-3083.2010.02443.x
21. Guinovart RM, Vicente A, Rovira C, Suñol M, González-Enseñat MA. Facial telangiectasia: an unusual manifestation of neonatal lupus erythematosus. Lupus. Apr 2012;21(5):552-5. doi:10.1177/0961203311430701
22. Silverman E. Neonatal lupus. In: Textbook of pediatric rheumatology, Cassidy JT, Petty RE (Eds), WB Saunders, Philadelphia 2001. p.456.
23. Savino F, Viola S, Tarasco V, Locatelli E, Ricagni A, Coppo P. Neonatal lupus erythematosus: a cutaneous cases based update. Ital J Pediatr. Jan 7 2016;42:1. doi:10.1186/s13052-015-0208-5
24. Baselga E, Torrelo A. Inflammatory and Purpuric Eruptions. Neonatal Dermatology. Copyright © 2008 Elsevier Inc. All rights reserved.; 2008:311-42.
25. Lau L, Krafchik B, Trebo MM, Weitzman S. Cutaneous Langerhans cell histiocytosis in children under one year. Pediatr Blood Cancer. Jan 2006;46(1):66-71. doi:10.1002/pbc.20479
26. Bonnekoh H, Butze M, Kallinich T, Kambe N, Kokolakis G, Krause K. Spectrum of Genetic Autoinflammatory Diseases Presenting with Cutaneous Symptoms. Acta Derm Venereol. Mar 25 2020;100(7):adv00091. doi:10.2340/00015555-3427
27. Peñate Y, Guillermo N, Rodríguez J, et al. Histopathologic characteristics of neonatal cutaneous lupus erythematosus: description of five cases and literature review. J Cutan Pathol. Jun 2009;36(6):660-7. doi:10.1111/j.1600-0560.2008.01136.x
28. Tahernia L, Alimadadi H, Tahghighi F, Amini Z, Ziaee V. Frequency and Type of Hepatic and Gastrointestinal Involvement in Juvenile Systemic Lupus Erythematosus. Autoimmune Dis. 2017;2017:8097273. doi:10.1155/2017/8097273
29. Zuppa AA, Riccardi R, Frezza S, et al. Neonatal lupus: Follow-up in infants with anti-SSA/Ro antibodies and review of the literature. Autoimmun Rev. Apr 2017;16(4):427-432. doi:10.1016/j.autrev.2017.02.010
30. Prendiville JS, Cabral DA, Poskitt KJ, Au S, Sargent MA. Central nervous system involvement in neonatal lupus erythematosus. Pediatr Dermatol. Jan-Feb 2003;20(1):60-7. doi:10.1046/j.1525-1470.2003.03014.x
31. Saini AG, Sankhyan N, Bhattad S, Vyas S, Saikia B, Singhi P. CNS vasculitis and stroke in neonatal lupus erythematosus: a case report and review of literature. Eur J Paediatr Neurol. May 2014;18(3):444-8. doi:10.1016/j.ejpn.2014.01.007
32. Capone C, Buyon JP, Friedman DM, Frishman WH. Cardiac manifestations of neonatal lupus: a review of autoantibody-associated congenital heart block and its impact in an adult population. Cardiol Rev. Mar-Apr 2012;20(2):72-6. doi:10.1097/CRD.0b013e31823c808b
33. Balmer C, Fasnacht M, Rahn M, Molinari L, Bauersfeld U. Long-term follow up of children with congenital complete atrioventricular block and the impact of pacemaker therapy. Europace. Oct 2002;4(4):345-9. doi:10.1053/eupc.2002.0266
34. Askanase AD, Friedman DM, Copel J, et al. Spectrum and progression of conduction abnormalities in infants born to mothers with anti-SSA/Ro-SSB/La antibodies. Lupus. 2002;11(3):145-51. doi:10.1191/0961203302lu173oa
35. Friedman DM, Rupel A, Glickstein J, Buyon JP. Congenital heart block in neonatal lupus: the pediatric cardiologist's perspective. Indian J Pediatr. Jun 2002;69(6):517-22. doi:10.1007/bf02722656
36. Nield LE, Silverman ED, Smallhorn JF, et al. Endocardial fibroelastosis associated with maternal anti-Ro and anti-La antibodies in the absence of atrioventricular block. J Am Coll Cardiol. Aug 21 2002;40(4):796-802. doi:10.1016/s0735-1097(02)02004-1
37. Hornberger LK, Al Rajaa N. Spectrum of cardiac involvement in neonatal lupus. Scand J Immunol. Sep 2010;72(3):189-97. doi:10.1111/j.1365-3083.2010.02437.x
38. Jaeggi ET, Hamilton RM, Silverman ED, Zamora SA, Hornberger LK. Outcome of children with fetal, neonatal or childhood diagnosis of isolated congenital atrioventricular block. A single institution's experience of 30 years. J Am Coll Cardiol. Jan 2 2002;39(1):130-7. doi:10.1016/s0735-1097(01)01697-7
39. Llanos C, Izmirly PM, Katholi M, et al. Recurrence rates of cardiac manifestations associated with neonatal lupus and maternal/fetal risk factors. Arthritis Rheum. Oct 2009;60(10):3091-7. doi:10.1002/art.24768
40. Eronen M, Sirèn MK, Ekblad H, Tikanoja T, Julkunen H, Paavilainen T. Short- and long-term outcome of children with congenital complete heart block diagnosed in utero or as a newborn. Pediatrics. Jul 2000;106(1 Pt 1):86-91. doi:10.1542/peds.106.1.86
41. Zhao H, Cuneo BF, Strasburger JF, Huhta JC, Gotteiner NL, Wakai RT. Electrophysiological characteristics of fetal atrioventricular block. J Am Coll Cardiol. Jan 1 2008;51(1):77-84. doi:10.1016/j.jacc.2007.06.060
42. Watson R, Kang JE, May M, Hudak M, Kickler T, Provost TT. Thrombocytopenia in the neonatal lupus syndrome. Arch Dermatol. Apr 1988;124(4):560-3.
43. Lee L. Neonatal lupus erythematosus and congenital heart block. In: Dyall-Smith D, Marks R, editors. Dermatology at the millennium: the proceedings of the 19th World Congress of Dermatology. London: The Parthenon Publishing Group, 1999: 273-7.
44. Kurien BT, Newland J, Paczkowski C, Moore KL, Scofield RH. Association of neutropenia in systemic lupus erythematosus (SLE) with anti-Ro and binding of an immunologically cross-reactive neutrophil membrane antigen. Clin Exp Immunol. Apr 2000;120(1):209-17. doi:10.1046/j.1365-2249.2000.01195.x
45. Lynn Cheng C, Galbraith S, Holland K. Congenital lupus erythematosus presenting at birth with widespread erosions, pancytopenia, and subsequent hepatobiliary disease. Pediatr Dermatol. Jan-Feb 2010;27(1):109-11. doi:10.1111/j.1525-1470.2009.01057.x
46. Wang L-J, Yang Y-H, Lin M-I, Chiang B-L, Mu S-C. Neonatal lupus erythematosus with recurrent pancytopenia: a case report. Journal of microbiology, immunology, and infection = Wei mian yu gan ran za zhi. 2002/12// 2002;35(4):262-264.
2. Chen CC, Lin KL, Chen CL, Wong AM, Huang JL. Central nervous system manifestations of neonatal lupus: a systematic review. Lupus. Dec 2013;22(14):1484-8. doi:10.1177/0961203313509294
3. 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. Jun 2009;68(6):828-35. doi:10.1136/ard.2008.088054
4. Shahian M, Khosravi A, Anbardar MH. Early cholestasis in neonatal lupus erythematosus. Ann Saudi Med. Jan-Feb 2011;31(1):80-2. doi:10.4103/0256-4947.70569
5. Boros CA, Spence D, Blaser S, Silverman ED. Hydrocephalus and macrocephaly: new manifestations of neonatal lupus erythematosus. Arthritis Rheum. Mar 15 2007;57(2):261-6. doi:10.1002/art.22543
6. Lee LA. Transient autoimmunity related to maternal autoantibodies: neonatal lupus. Autoimmun Rev. Apr 2005;4(4):207-13. doi:10.1016/j.autrev.2004.11.003
7. Izmirly PM, Saxena A, Sahl SK, et al. Assessment of fluorinated steroids to avert progression and mortality in anti-SSA/Ro-associated cardiac injury limited to the fetal conduction system. Ann Rheum Dis. Jun 2016;75(6):1161-5. doi:10.1136/annrheumdis-2015-208311
8. Izmirly PM, Saxena A, Kim MY, et al. Maternal and fetal factors associated with mortality and morbidity in a multi-racial/ethnic registry of anti-SSA/Ro-associated cardiac neonatal lupus. Circulation. Nov 1 2011;124(18):1927-35. doi:10.1161/circulationaha.111.033894
9. Cimaz R, Spence DL, Hornberger L, Silverman ED. Incidence and spectrum of neonatal lupus erythematosus: a prospective study of infants born to mothers with anti-Ro autoantibodies. J Pediatr. Jun 2003;142(6):678-83. doi:10.1067/mpd.2003.233
10. Vanoni F, Lava SAG, Fossali EF, et al. Neonatal Systemic Lupus Erythematosus Syndrome: a Comprehensive Review. Clin Rev Allergy Immunol. Dec 2017;53(3):469-476. doi:10.1007/s12016-017-8653-0
11. Strandberg L, Salomonsson S, Bremme K, Sonesson S, Wahren-Herlenius M. Ro52, Ro60 and La IgG autoantibody levels and Ro52 IgG subclass profiles longitudinally throughout pregnancy in congenital heart block risk pregnancies. Lupus. 2006;15(6):346-53. doi:10.1191/0961203306lu2309oa
12. Yoshimi R, Ueda A, Ozato K, Ishigatsubo Y. Clinical and pathological roles of Ro/SSA autoantibody system. Clin Dev Immunol. 2012;2012:606195. doi:10.1155/2012/606195
13. Gleicher N, Elkayam U. Preventing congenital neonatal heart block in offspring of mothers with anti-SSA/Ro and SSB/La antibodies: a review of published literature and registered clinical trials. Autoimmun Rev. Sep 2013;12(11):1039-45. doi:10.1016/j.autrev.2013.04.006
14. Provost TT, Watson R, Gammon WR, Radowsky M, Harley JB, Reichlin M. The neonatal lupus syndrome associated with U1RNP (nRNP) antibodies. N Engl J Med. Apr 30 1987;316(18):1135-8. doi:10.1056/nejm198704303161807
15. Sheth AP, Esterly NB, Ratoosh SL, Smith JP, Hebert AA, Silverman E. U1RNP positive neonatal lupus erythematosus: association with anti-La antibodies? Br J Dermatol. Apr 1995;132(4):520-6. doi:10.1111/j.1365-2133.1995.tb08705.x
16. Shiiya C, Ota M. Facial Rash, Fever, and Anemia in a Newborn. Jama. May 23 2017;317(20):2125-2126. doi:10.1001/jama.2017.3058
17. Lee LA. Neonatal lupus: clinical features and management. Paediatr Drugs. 2004;6(2):71-8. doi:10.2165/00148581-200406020-00001
18. Sommer LL, Manders SM. Seeing double: annular diaper rash in twins. Pediatr Dermatol. May-Jun 2015;32(3):410-3. doi:10.1111/pde.12508
19. See A, Wargon O, Lim A, Sugo E, Le K. Neonatal lupus erythematosus presenting as papules on the feet. Australas J Dermatol. Aug 2005;46(3):172-6. doi:10.1111/j.1440-0960.2005.00173.x
20. Silverman E, Jaeggi E. Non-cardiac manifestations of neonatal lupus erythematosus. Scand J Immunol. Sep 2010;72(3):223-5. doi:10.1111/j.1365-3083.2010.02443.x
21. Guinovart RM, Vicente A, Rovira C, Suñol M, González-Enseñat MA. Facial telangiectasia: an unusual manifestation of neonatal lupus erythematosus. Lupus. Apr 2012;21(5):552-5. doi:10.1177/0961203311430701
22. Silverman E. Neonatal lupus. In: Textbook of pediatric rheumatology, Cassidy JT, Petty RE (Eds), WB Saunders, Philadelphia 2001. p.456.
23. Savino F, Viola S, Tarasco V, Locatelli E, Ricagni A, Coppo P. Neonatal lupus erythematosus: a cutaneous cases based update. Ital J Pediatr. Jan 7 2016;42:1. doi:10.1186/s13052-015-0208-5
24. Baselga E, Torrelo A. Inflammatory and Purpuric Eruptions. Neonatal Dermatology. Copyright © 2008 Elsevier Inc. All rights reserved.; 2008:311-42.
25. Lau L, Krafchik B, Trebo MM, Weitzman S. Cutaneous Langerhans cell histiocytosis in children under one year. Pediatr Blood Cancer. Jan 2006;46(1):66-71. doi:10.1002/pbc.20479
26. Bonnekoh H, Butze M, Kallinich T, Kambe N, Kokolakis G, Krause K. Spectrum of Genetic Autoinflammatory Diseases Presenting with Cutaneous Symptoms. Acta Derm Venereol. Mar 25 2020;100(7):adv00091. doi:10.2340/00015555-3427
27. Peñate Y, Guillermo N, Rodríguez J, et al. Histopathologic characteristics of neonatal cutaneous lupus erythematosus: description of five cases and literature review. J Cutan Pathol. Jun 2009;36(6):660-7. doi:10.1111/j.1600-0560.2008.01136.x
28. Tahernia L, Alimadadi H, Tahghighi F, Amini Z, Ziaee V. Frequency and Type of Hepatic and Gastrointestinal Involvement in Juvenile Systemic Lupus Erythematosus. Autoimmune Dis. 2017;2017:8097273. doi:10.1155/2017/8097273
29. Zuppa AA, Riccardi R, Frezza S, et al. Neonatal lupus: Follow-up in infants with anti-SSA/Ro antibodies and review of the literature. Autoimmun Rev. Apr 2017;16(4):427-432. doi:10.1016/j.autrev.2017.02.010
30. Prendiville JS, Cabral DA, Poskitt KJ, Au S, Sargent MA. Central nervous system involvement in neonatal lupus erythematosus. Pediatr Dermatol. Jan-Feb 2003;20(1):60-7. doi:10.1046/j.1525-1470.2003.03014.x
31. Saini AG, Sankhyan N, Bhattad S, Vyas S, Saikia B, Singhi P. CNS vasculitis and stroke in neonatal lupus erythematosus: a case report and review of literature. Eur J Paediatr Neurol. May 2014;18(3):444-8. doi:10.1016/j.ejpn.2014.01.007
32. Capone C, Buyon JP, Friedman DM, Frishman WH. Cardiac manifestations of neonatal lupus: a review of autoantibody-associated congenital heart block and its impact in an adult population. Cardiol Rev. Mar-Apr 2012;20(2):72-6. doi:10.1097/CRD.0b013e31823c808b
33. Balmer C, Fasnacht M, Rahn M, Molinari L, Bauersfeld U. Long-term follow up of children with congenital complete atrioventricular block and the impact of pacemaker therapy. Europace. Oct 2002;4(4):345-9. doi:10.1053/eupc.2002.0266
34. Askanase AD, Friedman DM, Copel J, et al. Spectrum and progression of conduction abnormalities in infants born to mothers with anti-SSA/Ro-SSB/La antibodies. Lupus. 2002;11(3):145-51. doi:10.1191/0961203302lu173oa
35. Friedman DM, Rupel A, Glickstein J, Buyon JP. Congenital heart block in neonatal lupus: the pediatric cardiologist's perspective. Indian J Pediatr. Jun 2002;69(6):517-22. doi:10.1007/bf02722656
36. Nield LE, Silverman ED, Smallhorn JF, et al. Endocardial fibroelastosis associated with maternal anti-Ro and anti-La antibodies in the absence of atrioventricular block. J Am Coll Cardiol. Aug 21 2002;40(4):796-802. doi:10.1016/s0735-1097(02)02004-1
37. Hornberger LK, Al Rajaa N. Spectrum of cardiac involvement in neonatal lupus. Scand J Immunol. Sep 2010;72(3):189-97. doi:10.1111/j.1365-3083.2010.02437.x
38. Jaeggi ET, Hamilton RM, Silverman ED, Zamora SA, Hornberger LK. Outcome of children with fetal, neonatal or childhood diagnosis of isolated congenital atrioventricular block. A single institution's experience of 30 years. J Am Coll Cardiol. Jan 2 2002;39(1):130-7. doi:10.1016/s0735-1097(01)01697-7
39. Llanos C, Izmirly PM, Katholi M, et al. Recurrence rates of cardiac manifestations associated with neonatal lupus and maternal/fetal risk factors. Arthritis Rheum. Oct 2009;60(10):3091-7. doi:10.1002/art.24768
40. Eronen M, Sirèn MK, Ekblad H, Tikanoja T, Julkunen H, Paavilainen T. Short- and long-term outcome of children with congenital complete heart block diagnosed in utero or as a newborn. Pediatrics. Jul 2000;106(1 Pt 1):86-91. doi:10.1542/peds.106.1.86
41. Zhao H, Cuneo BF, Strasburger JF, Huhta JC, Gotteiner NL, Wakai RT. Electrophysiological characteristics of fetal atrioventricular block. J Am Coll Cardiol. Jan 1 2008;51(1):77-84. doi:10.1016/j.jacc.2007.06.060
42. Watson R, Kang JE, May M, Hudak M, Kickler T, Provost TT. Thrombocytopenia in the neonatal lupus syndrome. Arch Dermatol. Apr 1988;124(4):560-3.
43. Lee L. Neonatal lupus erythematosus and congenital heart block. In: Dyall-Smith D, Marks R, editors. Dermatology at the millennium: the proceedings of the 19th World Congress of Dermatology. London: The Parthenon Publishing Group, 1999: 273-7.
44. Kurien BT, Newland J, Paczkowski C, Moore KL, Scofield RH. Association of neutropenia in systemic lupus erythematosus (SLE) with anti-Ro and binding of an immunologically cross-reactive neutrophil membrane antigen. Clin Exp Immunol. Apr 2000;120(1):209-17. doi:10.1046/j.1365-2249.2000.01195.x
45. Lynn Cheng C, Galbraith S, Holland K. Congenital lupus erythematosus presenting at birth with widespread erosions, pancytopenia, and subsequent hepatobiliary disease. Pediatr Dermatol. Jan-Feb 2010;27(1):109-11. doi:10.1111/j.1525-1470.2009.01057.x
46. Wang L-J, Yang Y-H, Lin M-I, Chiang B-L, Mu S-C. Neonatal lupus erythematosus with recurrent pancytopenia: a case report. Journal of microbiology, immunology, and infection = Wei mian yu gan ran za zhi. 2002/12// 2002;35(4):262-264.
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Mar 13, 2023
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Bullock, T., Simonds, R., McBride, J., Irfan, M., & Young, S. (2023). Neonatal Lupus Erythematosus: A Case Report and Review of the Literature. SKIN The Journal of Cutaneous Medicine, 7(2), 649–656. https://doi.org/10.25251/skin.7.2.1
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