Hypergammaglobulinemic Purpura Occuring in a Female Delivered of an Infant Diagnosed with Neonatal Lupus Erythematosus.

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  • 新生児エリテマトーデス児を出産した高γグロブリン血症性紫斑

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Abstract

A 31-year-old pregnant female presented with an 8-month history of purpura on both legs. This condition worsened after becoming pregnant. A history of facial annular erythema at age 22 and purpura experienced after standing for long periods from age 28 was elicited. A physical examination revealed 2-3mm multiple erythema and purpura on both legs, intermingled with plaques of pigmentation. The titers of anti SS-A and SS-B antibodies were 30.8 and 138.4 on ELISA index, respectively, and the level of γ-globulin was 2.0g/dl. Rose-Bengal and fluorescein tests elicited positive results. A histological examination revealed predominantly dermal perivascular neutrophilic cellular infiltration with nuclear dust and extravasation of erythrocytes. Purpura and pigmentation improved with rest from exertion at about 9 months into pregnancy, but the pigmentation remained. The baby was diagnosed to have neonatal lupus erythematosus following the observation of annular erythema, a ventricular septal defect and high titers of anti SS-A and SS-B antibodies. These results indicate that the mechanism of maternal purpura formation may be an obstruction of the venous return at the inferior vena cava (due to pressure from the enlarged uterus), vasculitis, increased γ-globulin and hyperviscosity, and increased plasma volume. However, the patient was also diagnosed to have Sjoögren’s syndrome according to high titers of anti SS-A and SS-B antibodies. In addition, she was also suspected to have keratoconjunctivitis sicca. She thereafter delivered an infant with neonatal lupus erythematosus. In conclusion, the present case suggests that the measurement of anti SS-A and SS-B antibody titers is important, and therefore careful consideration should be given to cardiac disease of the fetus in pregnant woman demonstrating hypergammaglobulinemic purpura.

Journal

  • Nishi Nihon Hifuka

    Nishi Nihon Hifuka 64 (4), 412-415, 2002

    Western Division of Japanese Dermatological Association

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