Possible Discoid Lupus Erythematosus in Newborn Infant

McCUISTION, C. H.

doi:10.1001/archderm.1954.01540240088009

Cited by 131 publications

(16 citation statements)

References 2 publications

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“…Neonatal lupus erythematosus (NLE) is a rare syndrome, which was first described as a distinct clinical and pathological entity in 1954 by McCuistion & Schoch [2]. The potentially severe and permanent manifestation of NLE, the complete congential heart block (CCHB), was first recognized in 1945 by Plant & Steven [3].…”

Section: Introductionmentioning

confidence: 99%

Complete congenital heart block is associated with increased autoantibody titers against calreticulin

Orth

Dörner

Büschenfelde

et al. 1996

Eur J Clin Investigation

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Abstract. Complete congenital heart block (CCHB) is associated with anti-Ro/SS-A and anti-La/SS-B antibodies. Calreticulin, a calcium-binding, multifunctional protein of the endoplasmic reticulum with C-terminal KDEL-sequence, is not part of the Ro/SS-A ribonucleoprotein complex. In this study anti-calreticulin autoantibody responses in serum samples from 18 infants with CCHB, their mothers and in a control group of 11 antiRo/SS-A or anti-La/SS-B positive infants without heart block and their mothers were analysed. Specific enzymelinked immunosorbent assays were performed. Nine out of 18 sera with CCHB contained IgG anti-calreticulin antibodies. Four sera of those with IgG antibodies also had IgM antibodies. One serum contained anti-calreticulin IgM antibodies only. In the non-CCHB group two sera were positive for IgG and one serum was positive for IgM anti-calreticulin antibodies. Sera of healthy infants were negative both for anti-IgG and anti-IgM calreticulin antibodies. Calreticulin is involved in calcium storage and therefore anti-calreticulin antibodies might influence the development of CCHB. The new finding of IgM autoantibodies and the observed differences in antibody response in infants and mothers support the hypothesis of a fetally mediated and passively acquired autoimmune disease.

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Section: Introductionmentioning

confidence: 99%

Complete congenital heart block is associated with increased autoantibody titers against calreticulin

Orth

Dörner

Büschenfelde

et al. 1996

Eur J Clin Investigation

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“…However, it appears to be relatively common. The 31 cases known to us are unlike the 19 cases of other neonatal diseases (McCuistion and Schoch, 1954;Dias et al, 1958;Nathan and Snapper, 1958;Seip, 1960;Epstein and Litt, 1961;Johnson, 1963(cited by Reed et al, 1967Jackson, 1964;Hull et al, 1966;Reed et al, 1967 (2 cases); Cruveiller et al, 1970;Jones, 1971;Hontani et al, 1971;Goldberg and Diamond, 1973;Weiner, 1973 (2 cases cited by Goldberg and Diamond, 1973;Vonderheid et al, 1976);Soltani et al, 1974;Vonderheid et al, 1976 (2 cases)), and unlike the 4 cases in which the neonatal illness was diagnosed as SLE but in which there was no maternal illness suggestive of SLE, with abnormal serological tests in 2 (Nice, 1962;East and Lumpkin, 1969;Vonderheid et al, 1976 (2 cases)).…”

Section: Spectrum Of Neonatal Diseasementioning

confidence: 78%

Congenital complete heart block in the newborn associated with maternal systemic lupus erythematosus and other connective tissue disorders.

Hardy¹,

Solomon²,

Banwell³

et al. 1979

Archives of Disease in Childhood

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SUMMARY Four babies with complete heart block associated with maternal systemic lupus erythematosus (SLE) are described, together with a 5th baby whose mother had serological abnormalities only. One baby had a rapidly fatal outcome, one has required digoxin for heart failure, and the remaining 3 are asymptomatic but remain in complete heart block. Additional manifestations were present in 2 of them. The spectrum of neonatal abnormalities that may occur in association with maternal SLE and related connective tissue disorders is discussed, together with the possible causes and the prognosis. We conclude that congenital heart block is more common than had previously been appreciated.

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“…The role of transplacental transmission of a factor responsible for the development of neonatal LE was postulated in McCuistion and Schoch's report (14). However, in 1966, Beck et a1 (31) reported the transplacental passage of 7 .OS gamma globulin, the antinuclear antibody, from 3 mothers with SLE.…”

Section: Discussionmentioning

confidence: 98%

Neonatal lupus erythematosus occurring in one fraternal twin. Serologic and immunogenetic studies

Callen

Fowler

Kulick

et al. 1985

Arthritis & Rheumatism

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Neonatal lupus erythematosus (LE) is a syndrome that is manifested by LE skin lesions and/or congenital heart block, occurring in infants at, or shortly after, birth. The syndrome is believed to be caused by transplacental passage of an IgG antibody, usually the anti‐Ro (SS‐A) antibody, from the mother to the infant. Although the mother may have a connective tissue disease or may be healthy, the common characteristic is the presence of maternal circulating anti‐Ro antibody. It has been believed that the HLA determinants demonstrated in children who have neonatal LE were not a factor in the expression of the syndrome. We report the occurrence of neonatal LE, manifested by photosensitivity and discoid LE skin lesions, in one fraternal twin. HLA studies of this affected twin demonstrated the presence of DR3. Anti‐Ro antibody was present in the mother, but was not present in either child at 4 months post‐delivery. HLA determinants may be involved in the expression of disease in neonates who have been exposed to the anti‐Ro antibody. Furthermore, the presence of circulating antibodies in the unaffected twin causes us to question the assumption that the anti‐Ro antibody is the causative factor for the occurrence of tissue injury in children with neonatal LE.

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Possible Discoid Lupus Erythematosus in Newborn Infant

Cited by 131 publications

References 2 publications

Complete congenital heart block is associated with increased autoantibody titers against calreticulin

Complete congenital heart block is associated with increased autoantibody titers against calreticulin

Congenital complete heart block in the newborn associated with maternal systemic lupus erythematosus and other connective tissue disorders.

Neonatal lupus erythematosus occurring in one fraternal twin. Serologic and immunogenetic studies

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