Neonatal lupus erythematosus with cholestatic hepatitis

Sheng-Chieh Lin, Shyh-Dar Shyur, Li-Hsin Huang, Jiunn-Yi Wu, Hsun T. Chuo, Hung-Chang Lee

Research output: Contribution to journalArticle

8 Citations (Scopus)

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.
Original languageEnglish
Pages (from-to)131-134
Number of pages4
JournalJournal of Microbiology, Immunology and Infection
Volume37
Issue number2
Publication statusPublished - 2004
Externally publishedYes

Fingerprint

Hepatitis
Thrombocytopenia
Skin
Hyperbilirubinemia
Antinuclear Antibodies
Prednisolone
Transaminases
Jaundice
Bilirubin
Autoantibodies
Autoimmune Diseases
Anemia
Anti-Idiotypic Antibodies
Heart Diseases
Hemoglobins
Differential Diagnosis
Mothers
Antibodies
Neonatal Systemic lupus erythematosus
Therapeutics

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

Cite this

Lin, S-C., Shyur, S-D., Huang, L-H., Wu, J-Y., Chuo, H. T., & Lee, H-C. (2004). Neonatal lupus erythematosus with cholestatic hepatitis. Journal of Microbiology, Immunology and Infection, 37(2), 131-134.

Neonatal lupus erythematosus with cholestatic hepatitis. / Lin, Sheng-Chieh; Shyur, Shyh-Dar; Huang, Li-Hsin; Wu, Jiunn-Yi; Chuo, Hsun T.; Lee, Hung-Chang.

In: Journal of Microbiology, Immunology and Infection, Vol. 37, No. 2, 2004, p. 131-134.

Research output: Contribution to journalArticle

Lin, S-C, Shyur, S-D, Huang, L-H, Wu, J-Y, Chuo, HT & Lee, H-C 2004, 'Neonatal lupus erythematosus with cholestatic hepatitis', Journal of Microbiology, Immunology and Infection, vol. 37, no. 2, pp. 131-134.
Lin, Sheng-Chieh ; Shyur, Shyh-Dar ; Huang, Li-Hsin ; Wu, Jiunn-Yi ; Chuo, Hsun T. ; Lee, Hung-Chang. / Neonatal lupus erythematosus with cholestatic hepatitis. In: Journal of Microbiology, Immunology and Infection. 2004 ; Vol. 37, No. 2. pp. 131-134.
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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.",
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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",
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TY - JOUR

T1 - Neonatal lupus erythematosus with cholestatic hepatitis

AU - Lin, Sheng-Chieh

AU - Shyur, Shyh-Dar

AU - Huang, Li-Hsin

AU - Wu, Jiunn-Yi

AU - Chuo, Hsun T.

AU - Lee, Hung-Chang

N1 - 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

PY - 2004

Y1 - 2004

N2 - 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.

AB - 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.

KW - Cholestasis

KW - Hepatitis

KW - Lupus erythematosus

KW - Newborn infant

KW - aminotransferase

KW - antinuclear antibody

KW - autoantibody

KW - bilirubin

KW - hemoglobin

KW - maternal antibody

KW - prednisolone

KW - ribonucleoprotein antibody

KW - Ro antibody

KW - anemia

KW - article

KW - autoimmune disease

KW - case report

KW - cholestatic hepatitis

KW - clinical feature

KW - congenital heart block

KW - differential diagnosis

KW - disease association

KW - fetomaternal transfusion

KW - heart disease

KW - hematologic disease

KW - hepatitis

KW - human

KW - hyperbilirubinemia

KW - infection

KW - jaundice

KW - lupus erythematosus

KW - male

KW - metabolic disorder

KW - newborn

KW - newborn disease

KW - newborn lupus erythematosus

KW - skin lupus erythematosus

KW - thrombocytopenia

KW - Anemia

KW - Antibodies, Antinuclear

KW - Autoantibodies

KW - Autoantigens

KW - Humans

KW - Infant

KW - Infant, Newborn

KW - Lupus Erythematosus, Systemic

KW - Male

KW - Prednisolone

KW - Ribonucleoproteins

KW - Thrombocytopenia

KW - Transaminases

M3 - Article

VL - 37

SP - 131

EP - 134

JO - Journal of Microbiology, Immunology and Infection

JF - Journal of Microbiology, Immunology and Infection

SN - 0253-2662

IS - 2

ER -