Bilateral adrenal hemorrhage during pregnancy and puerperium has been associated with preeclampsia, sepsis, or blood transfusion reactions. We describe a patient with recurrent fetal loss and antiphospholipid antibodies in whom acute adrenal hemorrhage and adrenal insufficiency developed after an uncomplicated cesarean delivery. This is the second case in which a patient recovered adrenal function after hemorrhage.

A 30-year-old woman with a history of one ectopic pregnancy and three spontaneous abortions had an uncomplicated cesarean delivery. She had no history or physical findings to suggest a connective tissue disorder. Eight days later, she was hospitalized with nausea, vomiting, and severe bilateral costovertebral angle pain. She was afebrile, normotensive, and without orthostasis. Laboratory findings included normal electrolyte levels and complete blood count. A computed tomographic scan of the abdomen showed bilateral adrenal enlargement consistent with adrenal hemorrhage.

A short corticotropin-stimulation test showed baseline and 60-minute cortisol values of 12.8 µg/dL and 11.9 µg/dL, respectively (normal 60-minute value, >20 µg/dL). A baseline adrenocorticotropin hormone level was 1073 ng/L (normal, <56 ng/L). The abdominal pain was promptly relieved with intravenously administered hydrocortisone. Further testing showed a normal activated partial thromboplastin time of 28 seconds (normal, 27 to 40 seconds), a negative VDRL test, and an absence of antinuclear antibodies. A test for IgM antiphospholipid antibody (Metpath, Horsham, Pennsylvania) showed titers of 200 (normal, <100). Three months later, the scan showed normal adrenal glands. Seven months later, a short corticotropin stimulation test showed baseline and 60-minute cortisol values of 12.3 µg/dL and 25.7 µg/dL, respectively. The baseline adrenocorticotropin hormone level was 53.0 ng/dL at that time. Anticardiolipin IgG and IgM antibodies by enzyme immunoassay were negative 1.5 years later.

Only one other recent report [1] describes recovery of adrenal function after bilateral adrenal hemorrhage. The authors appropriately suggested that adrenal function be re-evaluated periodically in such patients.

Our patient had an uncomplicated delivery and did not appear to have any postoperative risk factors for adrenal hemorrhage. We propose that she had the primary antiphospholipid syndrome as a risk factor for adrenal hemorrhage. Although anticardiolipin antibody titers 1.5 years later were negative, these titers are known to fluctuate [2]. Two other patients with primary antiphospholipid syndrome [3, 4] were found to have adrenal insufficiency after being placed on anticoagulants for pulmonary emboli. Rao and colleagues [5] reported a 20-year-old patient with lupus anticoagulant who had bilateral adrenal hemorrhage not associated with anticoagulation or acute concomitant illness. The possible association between adrenal hemorrhage and the primary antiphospholipid syndrome requires further study.