Antiphospholipid syndrome and other autoimmune diseases associated with chronic intervillositis.

Abstract

Chronic intervillositis of unknown etiology (CIUE) is characterized by an intervillous infiltrate of mononuclear cells and a high recurrence rate of adverse obstetrical outcomes. The aim was to describe obstetrical history in patients with at least one event characterized by CIUE, and the possible impact of systematic investigation of an underlying autoimmune disease on the obstetrical outcome of subsequent pregnancies. We retrospectively reviewed all pregnancies in patients having experienced at least one adverse obstetric outcome associated with chronic intervillositis of unknown etiology diagnosed by placental histological analysis between 2004 and 2011 in our university hospital. For each patient, data pertaining to obstetrical history, treatments during pregnancies, the results of systematic investigation of an underlying autoimmune disease, and treatments as well as obstetrical outcome in subsequent pregnancies, were collected. Twelve patients with 38 pregnancies were included [median age 30 (22; 40 years)]. Autoimmune disease or autoimmune antibodies (AID group) were found in 7/12 patients: primary antiphospholipid syndrome (APS) (n = 4), Sjögren's syndrome (n = 1), pernicious anemia (n = 1) and celiac disease (n = 1). When comparing pregnancies of patients with and without AID, there was no difference with regard to the type of obstetrical events or live-born babies, in spite of appropriate treatment. Corticosteroids (prednisone 10 mg/day) were used in only 2 cases with AID (Sjögren's syndrome and APS; n = 1 each), and these 2 pregnancies resulted in live-born babies. This study shows that the immunological assessment in patients with CIUE raises the possibility of a specific severity when AID or obstetrical APS is associated with CIUE, since conventional treatment did not improve obstetrical outcome in these patients as compared to those without autoimmune diseases. The benefit of immunosuppressant agents in this subset of patients needs further evaluation.