Abstract
Background: Off-label use of intravenous immunoglobulin has been reported for the treatment of recurrent spontaneous pregnancy loss and selected cases of multiple sclerosis. However, the use of subcutaneous immunoglobulin has not been reported in these settings. Methods and results: We report on tolerability and clinical efficacy of subcutaneous immunoglobulin therapy in a woman with the unusual association of IgG4 subclass deficiency, C4 hypocomplementemia, localized scleroderma, unexplained spontaneous pregnancy loss and multiple sclerosis. Weekly infusions of subcutaneous immunoglobulin were self-administered at home (6 grams/week, Hizentra®) during the next pregnancy after abortion. Interferon beta-1a for the treatment of relapsing-remitting multiple sclerosis was stopped before pregnancy. During pregnancy only subcutaneous immunoglobulin was administered. The patient was found to have 4% of CD3+CD16/CD56+ (Natural Killer (NK) /T cells). The patient delivered a healthy baby (Apgar score 9 at 5 minutes) at 37 weeks gestation. During pregnancy and 3 months follow-up after delivery no multiple sclerosis flare-ups were observed. NK-T cell percentages remained stable. Conclusions: Our observation indicates that subcutaneous immunoglobulin therapy was well tolerated and efficacious in a selected case of unexplained abortion and multiple sclerosis.
Highlights
Recurrent pregnancy loss is defined as two or more pregnancy losses before gestational week 20
We report on the tolerability and clinical efficacy of subcutaneous immunoglobulin therapy in a woman with the unusual association of IgG4 subclass deficiency, multiple sclerosis and unexplained spontaneous pregnancy loss
A cut-off value of 3.75% for the increased percentage of NK-T cells in the peripheral blood has been suggested as a potential biomarker of pregnancy failure [6]
Summary
Recurrent pregnancy loss is defined as two or more pregnancy losses before gestational week 20. Immunological abnormalities are hypothesized to play a role in recurrent miscarriage [1]. Distinct types of immune-based therapies have been evaluated including intravenous immunoglobulins [2]. On the other hand pregnancy is associated with stabilization of autoimmune diseases, such as multiple sclerosis [3]. Pregnancy in women with multiple sclerosis is not generally considered high risk, there are some associated therapeutic challenges. Previous data suggest that pregnancy exposure to interferon-beta might result in lower mean birth weight and preterm birth [4] while other studies suggest the safety of this intervention [5]. We report on the tolerability and clinical efficacy of subcutaneous immunoglobulin therapy in a woman with the unusual association of IgG4 subclass deficiency, multiple sclerosis and unexplained spontaneous pregnancy loss
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