This report discusses our experience with two patients who had unilateral adrenalectomy during pregnancy as treatment for Cushing's syndrome secondary to an adrenal adenoma. Previously only five patients with this clinical problem who underwent unilateral adrenalectomy during pregnancy had been reported. We have reviewed the world literature on Cushing's syndrome in pregnancy secondary to an adrenal adenoma. A total of 19 patients who had unilateral adrenalectomy for this problem after the completion of pregnancy were identified. The review of world literature and the two patients who are the subject of this report were the basis of our analysis of fetal death, neonatal complications, and maternal complications in seven pregnancies during which unilateral adrenalectomy was performed (group 1) compared to the 19 pregnancies that were associated with unilateral adrenalectomy at the completion of pregnancy (group 2). Of the seven pregnancies in group 1, one fetal death and no neonatal complications occurred, but fetal death and neonatal complications occurred in 12 of the 19 pregnancies in group 2. Four of the seven mothers in group 1 had complications; 16 of the 19 mothers in group 2 had complications. This study suggests that adrenalectomy during pregnancy should be considered as a therapeutic option in the management of Cushing's syndrome secondary to an adrenal cortical adenoma.