@article{3e3b2c5f9e894545854ad687b8f7bade,
title = "Neonatal lupus erythematosus with cholestatic hepatitis",
abstract = "Neonatal lupus erythematosus is an uncommon passive autoimmune disease in which there is transplacental passage of anti-Ro/SSA and/or anti-La/SSB or anti-U1RNP maternal autoantibodies. Its common clinical manifestations include cardiac disease, notably congenital heart block, cutaneous lupus lesions, and hematologic problems. During the past decade, it has become clear that hepatobiliary disease may also occur as a manifestation of neonatal lupus erythematosus. We report a case of neonatal lupus erythematosus in a male infant who had lupus hepatitis with jaundice in addition to cutaneous lupus, anemia, and thrombocytopenia. Other diseases in the differential diagnosis of conjugated hyperbilirubinemia, including metabolic, infectious, and inherited anatomic conditions were all ruled out. The infant had a high titer of antinuclear antibodies (titer 1:640) with a speckled pattern, anti-Ro/SSA and anti-La/SSB antibodies, and no anti-dsDNA antibodies. Treatment with prednisolone (2 mg/kg/day) for 14 days resulted in dramatic improvement of the thrombocytopenia. Hemoglobin and bilirubin returned to normal 2 months later, and transaminases were normal by 10 months of age.",
keywords = "Cholestasis, Hepatitis, Lupus erythematosus, Newborn infant, aminotransferase, antinuclear antibody, autoantibody, bilirubin, hemoglobin, maternal antibody, prednisolone, ribonucleoprotein antibody, Ro antibody, anemia, article, autoimmune disease, case report, cholestatic hepatitis, clinical feature, congenital heart block, differential diagnosis, disease association, fetomaternal transfusion, heart disease, hematologic disease, hepatitis, human, hyperbilirubinemia, infection, jaundice, lupus erythematosus, male, metabolic disorder, newborn, newborn disease, newborn lupus erythematosus, skin lupus erythematosus, thrombocytopenia, Anemia, Antibodies, Antinuclear, Autoantibodies, Autoantigens, Humans, Infant, Infant, Newborn, Lupus Erythematosus, Systemic, Male, Prednisolone, Ribonucleoproteins, Thrombocytopenia, Transaminases",
author = "Sheng-Chieh Lin and Shyh-Dar Shyur and Li-Hsin Huang and Jiunn-Yi Wu and Chuo, {Hsun T.} and Hung-Chang Lee",
note = "被引用次數：7 Export Date: 7 April 2016 CODEN: JMIIF 通訊地址: Shyur, S.-D.; Department of Pediatrics, Mackay Memorial Hospital, 92, Section 2, Chung-Shan North Road, Taipei, 104, Taiwan 化學物質/CAS: aminotransferase, 9031-66-7; bilirubin, 18422-02-1, 635-65-4; hemoglobin, 9008-02-0; prednisolone, 50-24-8; Antibodies, Antinuclear; Autoantibodies; Autoantigens; Prednisolone, 50-24-8; Ribonucleoproteins; SS-B antigen; Transaminases, EC 2.6.1.- 參考文獻: Lee, L.A., Sokol, R.J., Buyon, J.P., Hepatobiliary disease in neonatal lupus: Prevalence and clinical characteristics in cases enrolled in a national registry (2002) Pediatrics, 109, pp. E11-E14; Burch, J.M., Lee, L.A., Weston, W.L., Neonatal lupus erythematosus (2002) Dermatol Nurs, 14, pp. 157-160; Brucato, A., Buyon, J.P., Horsfall, A.C., Lee, L.A., Reichlin, M., Fourth international workshop on neonatal lupus syndromes and the Ro/SSA-La/SSB system (1999) Clin Exp Rheumatol, 17, pp. 30-36; Lee, L.A., Neonatal lupus erythematosus (1993) J Invest Dermatol, 100 (SUPPL.), pp. S9-13; Le Thi Houng, D., Wechsler, B., Piette, J.C., Bletry, O., Godean, P., Pregnancy and its outcome in systemic lupus erythematosus (1994) QJM, 87, pp. 721-729; Kim, J., Smith, K.J., Skelton, H., Neonatal lupus erythematosus: Factors which may lead to clinical disease in the foetus even in the absence of disease in the mother (2001) Clin Exp Dermatol, 26, pp. 423-426; McCuistion, C.H., Schoch, E.P., Possible discoid lupus erythematosus in a newborn infant (1954) Arch Dermatol, 70, pp. 782-785; Rosh, J.R., Silverman, E.D., Groisman, G., Dolgin, S., LeLeiko, N.S., Intrahepatic cholestasis in neonatal lupus erythematosus (1993) J Pediatric Gastroenterol Nutr, 17, pp. 310-312; Weston, W.L., Morelli, J.G., Lee, L.A., The clinical spectrum of anti-Ro-positive cutaneous neonatal lupus erythematosus (1999) J Am Acad Dermatol, 40, pp. 675-681; Furukawa, F., Antinuclear antibody-keratinocyte interactions in photosensitive cutaneous lupus erythematosus (1999) Histol Histopathol, 14, pp. 627-633; Watson, R., Kang, J.E., May, M., Hudak, M., Kickler, T., Provost, T.T., Thrombocytopenia in the neonatal lupus syndrome (1988) Arch Dermatol, 124, pp. 560-563; Laxer, R.M., Robert, E.A., Gross, K.R., Britton, J.R., Cutz, E., Dimick, J., Liver disease in neonatal lupus erythematosus (1990) J Pediatr, 116, pp. 238-242; Lee, L.A., Reichlin, M., Ruyle, S.Z., Weston, W.L., Neonatal lupus liver disease (1993) Lupus, 2, pp. 333-338; Long, S.S., Moyer, V.A., Jobe, A.H., Welch, T.R., Daniels, S.R., Wilmott, R.W., The Editors' perspectives: Liver disease in neonatal lupus erythematosus (2002) J Pediatr, 140, pp. 1a-4a; Silverman, E.D., Neonatal lupus erythematosus (2001) Textbook of Pediatric Rheumatology. 4th Ed., pp. 450-463. , Cassidy JT, Petty RE, eds. Philadelphia: WB Saunders; Evans, N., Gaskin, K., Liver disease in association with neonatal lupus erythematosus (1993) J Paediatr Child Health, 29, pp. 478-480; Seip, M., Systemic lupus erythematosus in pregnancy with hemolytic anemia, leukopenia and thrombocytopenia in the mother and her newborn infant (1960) Arch Dis Child, 35, pp. 364-366; Hariharan, D., Manno, C.S., Seri, I., Neonatal lupus erythematosus with microvascular hemolysis (2000) J Pediatr Hematol Oncol, 22, pp. 351-354",
year = "2004",
language = "English",
volume = "37",
pages = "131--134",
journal = "Journal of Microbiology, Immunology and Infection",
issn = "1684-1182",
publisher = "Lippincott Williams & Wilkins Asia",
number = "2",
}
